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PMC11697044
A 68-year-old man was referred to the vascular surgery department for evaluation of an EIA aneurysm incidentally found on a screening magnetic resonance imaging after an elevated prostate-specific antigen on routine screening laboratory tests. Medical history was significant for type 2 diabetes mellitus, hypertension, hyperlipidemia, and coronary artery disease without a history of myocardial infarction or preventative intervention. He presented to the office and was evaluated for symptoms; he reported none. There was no history of trauma, infectious etiology, or prior vascular access. The patient denied any history of smoking, cycling or extreme sporting, or family history of aneurysms. Blood cultures were negative, and leukocytes were within normal limits. Computed tomography angiogram of the chest, abdomen, and pelvis with runoff revealed an isolated right sided saccular 2.6-cm EIA aneurysm above the inguinal ligament, with no extension proximally or distally . There was no aneurysmal or major atherosclerotic disease in the abdominal aorta or distal arterial vessels and there were no signs of disease in the contralateral iliac arteries. Based on discussion with the vascular surgery team, the patient was given the options of endovascular vs open surgery. Using shared decision-making, an endovascular approach was chosen to treat the isolated EIA aneurysm, and the patient provided consent. Fig 1 Preoperative computed tomography angiography showing an isolated aneurysm of the suprainguinal right external iliac artery ( EIA ). ( A ) Sagittal. ( B ) Axial. ( C ) Coronal with measurement. The operation was performed with the patient under general anesthesia. An 8F short 25 cm sheath (Terumo Medical Co, Tokyo, Japan) was placed percutaneously at the left femoral artery, and the Omni Flush Soft-Vu Angiographic Catheter (Angiodynamics, Latham, NY) was advanced and positioned into the infrarenal abdominal aorta. An aortogram was captured to visualize the iliac arteries with an oblique view to visualize the right hypogastric takeoff. A widely patent bilateral iliac system was visualized, and a large 2.6-m aneurysm was identified 4 cm above the femoral bifurcation. The Omni Flush over a floppy Glidewire (Terumo Medical Corp., Somerset, NJ) was advanced across the iliac bifurcation, beyond the aneurysm sac, and further down into the right superficial femoral artery. The short 8F sheath was exchanged for an 8F 45 cm Ansel Sheath (Cook Medical, Bloomington, IN) over a J-tipped Stiff Amplatz Wire (Boston Scientific, Natick, MA), and advanced to the mid-right EIA just proximal to the aneurysm. After heparinizing the patient and measuring the native vessel for optimal graft selection, the 9 mm × 10 cm Viabahn stent graft (W. L. Gore & Associates, Flagstaff, AZ) was ultimately selected and carefully deployed in a distal to proximal fashion. The entirety of the aneurysmal sac was covered while ensuring maintaining patency of the common femoral artery distally and hypogastric artery proximally. Final angiography demonstrated widely patent right EIA and widely patent femoral bifurcation with complete exclusion of the EIA aneurysm. Given that the stent graft appeared to have an excellent seal, it was decided not to post-dilate with balloon angioplasty. Fig 2 Intraoperative angiography. ( A ) Preoperative aortogram. ( B ) Preoperative iliac angiogram. ( C ) Completion angiogram after stent graft deployment. ( D ) Reconstructed three-dimensional image of 2-week follow-up computed tomography scan showing completely excluded aneurysm with patent stent graft in suprainguinal right external iliac artery ( EIA ). The patient was discharged home on postoperative day 1 without complication on a regimen of aspirin only, indefinitely. On outpatient review at 2 weeks, the patient was well. No difference in peripheral pulse examination was found. The patient was asymptomatic preoperatively and remained symptom free at the 2-week follow-up. Postoperative computed tomography angiography demonstrated an excluded aneurysm sac with a good apposition of the stent graft with no evidence of endoleaks or stent graft-related complications . We plan to perform annual ultrasound surveillance of the stent graft. Common and internal iliac artery aneurysms have multifactorial pathogeneses that are nearly identical to that of abdominal aortic aneurysms, as seen by their histological similarities. The particular rarity of aneurysms involving the EIA can be attributed to the unique lamellar architecture and biomechanical properties of the external iliac arterial walls, particularly in the tunica media. 7 Distinct from the more proximal aortoiliac segments, external iliac arteries possess a more structured and layered lamellar architecture, as well as a higher elastin-to-collagen ratio, 8 which allows them to withstand higher hemodynamic stresses and accommodate higher pressures, thus reducing the susceptibility to wall weakening and aneurysmal dilation. Isolated iliac artery aneurysms, without any other identifiable aortoiliac or peripheral vascular disease, have been described in multiple investigations to be a rare pathology. Silver et al 9 in 1967 performed a chart review of patients with arterial aneurysms affecting the aortic or iliac artery systems and found 571 patients with abdominal aortic aneurysms and only 11 patients with isolated iliac artery aneurysms, a relative frequency of 1.9%. Later in 1983, McCready et al 10 reported the frequency of isolated iliac artery aneurysms of 0.9% and provided one of the only anatomical frequency distributions amongst isolated aneurysms of the iliac artery system: 90% of all isolated iliac artery aneurysms affect the common iliac artery solely, whereas <1% affect the EIA solely. Finally, in 1989 Brunkwall et al 5 performed the largest investigation on isolated iliac artery aneurysms, reporting 13 cases during the 15-year compilation of autopsy and operating records in Malmo Sweden, population 230,000. They found only one isolated EIA aneurysm in that same study. 5 Regardless of how rare they are, many investigations have demonstrated that isolated iliac artery aneurysm are associated with a high risk of rupture and mortality, with rates of rupture between 14% and 75%. 2 , 11 , 12 The high mortality rate is postulated to be due to the lack of inclusion of iliac artery aneurysms in a differential diagnosis of pelvic conditions, partly owing to their rarity. Also, owing to the nature and location of the pathology, they are difficult to detect on physical exam until they are at a size when they are at risk for a morbid rupture. 3 McCready et al described that 78% of the patients in their study presented asymptomatically with their iliac artery aneurysm, similar to the patient described in this case study. Few cases have been reported describing isolated aneurysms of the EIA ( Table ). The first case report in 1952 described a patient who presented symptomatically with abdominal and lower limb pain. Surgical exploration revealed an EIA aneurysm that had ruptured. 13 The next three case reports published between 1986 and 2009 described iliac artery aneurysms involving the EIA that were found histologically to be due to cystic medial necrosis. 14 , 15 , 16 More recently, in 2019 two cases were presented with a 65-year-old symptomatic patient with left lower limb edema and a 55-year-old symptomatic patient with intermittent left thigh pain associated with paresthesias, both ipsilateral to the isolated EIA aneurysm. 17 , 18 Finally in 2020, there was another case report, similar to Crivello's, describing a symptomatic isolated EIA aneurysm associated with cystic medial necrosis. 19 In the present report, we have presented the case of a 68-year-old man who was completely asymptomatic, with an incidental finding of an EIA aneurysm. Until the presentation of our case, a search of PubMed found only seven reported cases of isolated EIA aneurysm, none of which were completely asymptomatic or repaired endovascularly. Table Reported cases of isolated external iliac artery ( EIA ) aneurysm Author Age, years Sex Size, cm Year Priddle et al 13 29 Female 4.0 1952 Crivello et al 14 27 Male Not available 1986 Mohan et al 15 66 Male 11.0 1997 Kato et al 16 78 Female 4.0 2009 Van de Luijtgaarden et al 17 65 Male 3.5 2019 Hussain et al 18 55 Male 7.0 2019 Chatzantonis et al 19 51 Male 2.0 2020 Current case 68 Male 2.6 2023 With this case report, we hope to stimulate a discussion on when to intervene on these rare pathologies. Although we have guidelines, based on large sample studies, for when to fix common or internal iliac artery aneurysms, the rarity of isolated EIA aneurysms leaves vascular surgeons without clear directions on when to fix them, especially when they are asymptomatic. Of the different publications reporting sizes of isolated EIA aneurysms, we found that most were repaired between 2 and 4 cm, mostly in men approximately 60 years old. Until clearer, large sample studies are performed, we recommend early repair of these aneurysms (diameter ≥2 cm) owing to the risk of rupture or symptoms seen in prior publications. Additionally, we now have the availability of minimally invasive interventions, with endografts that are able to be surveilled postoperatively with ultrasound examination. None. None.
Clinical case
clinical
en
0.999996
PMC11697044
A 68-year-old man was referred to the vascular surgery department for evaluation of an EIA aneurysm incidentally found on a screening magnetic resonance imaging after an elevated prostate-specific antigen on routine screening laboratory tests. Medical history was significant for type 2 diabetes mellitus, hypertension, hyperlipidemia, and coronary artery disease without a history of myocardial infarction or preventative intervention. He presented to the office and was evaluated for symptoms; he reported none. There was no history of trauma, infectious etiology, or prior vascular access. The patient denied any history of smoking, cycling or extreme sporting, or family history of aneurysms. Blood cultures were negative, and leukocytes were within normal limits. Computed tomography angiogram of the chest, abdomen, and pelvis with runoff revealed an isolated right sided saccular 2.6-cm EIA aneurysm above the inguinal ligament, with no extension proximally or distally . There was no aneurysmal or major atherosclerotic disease in the abdominal aorta or distal arterial vessels and there were no signs of disease in the contralateral iliac arteries. Based on discussion with the vascular surgery team, the patient was given the options of endovascular vs open surgery. Using shared decision-making, an endovascular approach was chosen to treat the isolated EIA aneurysm, and the patient provided consent. Fig 1 Preoperative computed tomography angiography showing an isolated aneurysm of the suprainguinal right external iliac artery ( EIA ). ( A ) Sagittal. ( B ) Axial. ( C ) Coronal with measurement. The operation was performed with the patient under general anesthesia. An 8F short 25 cm sheath (Terumo Medical Co, Tokyo, Japan) was placed percutaneously at the left femoral artery, and the Omni Flush Soft-Vu Angiographic Catheter (Angiodynamics, Latham, NY) was advanced and positioned into the infrarenal abdominal aorta. An aortogram was captured to visualize the iliac arteries with an oblique view to visualize the right hypogastric takeoff. A widely patent bilateral iliac system was visualized, and a large 2.6-m aneurysm was identified 4 cm above the femoral bifurcation. The Omni Flush over a floppy Glidewire (Terumo Medical Corp., Somerset, NJ) was advanced across the iliac bifurcation, beyond the aneurysm sac, and further down into the right superficial femoral artery. The short 8F sheath was exchanged for an 8F 45 cm Ansel Sheath (Cook Medical, Bloomington, IN) over a J-tipped Stiff Amplatz Wire (Boston Scientific, Natick, MA), and advanced to the mid-right EIA just proximal to the aneurysm. After heparinizing the patient and measuring the native vessel for optimal graft selection, the 9 mm × 10 cm Viabahn stent graft (W. L. Gore & Associates, Flagstaff, AZ) was ultimately selected and carefully deployed in a distal to proximal fashion. The entirety of the aneurysmal sac was covered while ensuring maintaining patency of the common femoral artery distally and hypogastric artery proximally. Final angiography demonstrated widely patent right EIA and widely patent femoral bifurcation with complete exclusion of the EIA aneurysm. Given that the stent graft appeared to have an excellent seal, it was decided not to post-dilate with balloon angioplasty. Fig 2 Intraoperative angiography. ( A ) Preoperative aortogram. ( B ) Preoperative iliac angiogram. ( C ) Completion angiogram after stent graft deployment. ( D ) Reconstructed three-dimensional image of 2-week follow-up computed tomography scan showing completely excluded aneurysm with patent stent graft in suprainguinal right external iliac artery ( EIA ). The patient was discharged home on postoperative day 1 without complication on a regimen of aspirin only, indefinitely. On outpatient review at 2 weeks, the patient was well. No difference in peripheral pulse examination was found. The patient was asymptomatic preoperatively and remained symptom free at the 2-week follow-up. Postoperative computed tomography angiography demonstrated an excluded aneurysm sac with a good apposition of the stent graft with no evidence of endoleaks or stent graft-related complications . We plan to perform annual ultrasound surveillance of the stent graft. Common and internal iliac artery aneurysms have multifactorial pathogeneses that are nearly identical to that of abdominal aortic aneurysms, as seen by their histological similarities. The particular rarity of aneurysms involving the EIA can be attributed to the unique lamellar architecture and biomechanical properties of the external iliac arterial walls, particularly in the tunica media. 7 Distinct from the more proximal aortoiliac segments, external iliac arteries possess a more structured and layered lamellar architecture, as well as a higher elastin-to-collagen ratio, 8 which allows them to withstand higher hemodynamic stresses and accommodate higher pressures, thus reducing the susceptibility to wall weakening and aneurysmal dilation. Isolated iliac artery aneurysms, without any other identifiable aortoiliac or peripheral vascular disease, have been described in multiple investigations to be a rare pathology. Silver et al 9 in 1967 performed a chart review of patients with arterial aneurysms affecting the aortic or iliac artery systems and found 571 patients with abdominal aortic aneurysms and only 11 patients with isolated iliac artery aneurysms, a relative frequency of 1.9%. Later in 1983, McCready et al 10 reported the frequency of isolated iliac artery aneurysms of 0.9% and provided one of the only anatomical frequency distributions amongst isolated aneurysms of the iliac artery system: 90% of all isolated iliac artery aneurysms affect the common iliac artery solely, whereas <1% affect the EIA solely. Finally, in 1989 Brunkwall et al 5 performed the largest investigation on isolated iliac artery aneurysms, reporting 13 cases during the 15-year compilation of autopsy and operating records in Malmo Sweden, population 230,000. They found only one isolated EIA aneurysm in that same study. 5 Regardless of how rare they are, many investigations have demonstrated that isolated iliac artery aneurysm are associated with a high risk of rupture and mortality, with rates of rupture between 14% and 75%. 2 , 11 , 12 The high mortality rate is postulated to be due to the lack of inclusion of iliac artery aneurysms in a differential diagnosis of pelvic conditions, partly owing to their rarity. Also, owing to the nature and location of the pathology, they are difficult to detect on physical exam until they are at a size when they are at risk for a morbid rupture. 3 McCready et al described that 78% of the patients in their study presented asymptomatically with their iliac artery aneurysm, similar to the patient described in this case study. Few cases have been reported describing isolated aneurysms of the EIA ( Table ). The first case report in 1952 described a patient who presented symptomatically with abdominal and lower limb pain. Surgical exploration revealed an EIA aneurysm that had ruptured. 13 The next three case reports published between 1986 and 2009 described iliac artery aneurysms involving the EIA that were found histologically to be due to cystic medial necrosis. 14 , 15 , 16 More recently, in 2019 two cases were presented with a 65-year-old symptomatic patient with left lower limb edema and a 55-year-old symptomatic patient with intermittent left thigh pain associated with paresthesias, both ipsilateral to the isolated EIA aneurysm. 17 , 18 Finally in 2020, there was another case report, similar to Crivello's, describing a symptomatic isolated EIA aneurysm associated with cystic medial necrosis. 19 In the present report, we have presented the case of a 68-year-old man who was completely asymptomatic, with an incidental finding of an EIA aneurysm. Until the presentation of our case, a search of PubMed found only seven reported cases of isolated EIA aneurysm, none of which were completely asymptomatic or repaired endovascularly. Table Reported cases of isolated external iliac artery ( EIA ) aneurysm Author Age, years Sex Size, cm Year Priddle et al 13 29 Female 4.0 1952 Crivello et al 14 27 Male Not available 1986 Mohan et al 15 66 Male 11.0 1997 Kato et al 16 78 Female 4.0 2009 Van de Luijtgaarden et al 17 65 Male 3.5 2019 Hussain et al 18 55 Male 7.0 2019 Chatzantonis et al 19 51 Male 2.0 2020 Current case 68 Male 2.6 2023 With this case report, we hope to stimulate a discussion on when to intervene on these rare pathologies. Although we have guidelines, based on large sample studies, for when to fix common or internal iliac artery aneurysms, the rarity of isolated EIA aneurysms leaves vascular surgeons without clear directions on when to fix them, especially when they are asymptomatic. Of the different publications reporting sizes of isolated EIA aneurysms, we found that most were repaired between 2 and 4 cm, mostly in men approximately 60 years old. Until clearer, large sample studies are performed, we recommend early repair of these aneurysms (diameter ≥2 cm) owing to the risk of rupture or symptoms seen in prior publications. Additionally, we now have the availability of minimally invasive interventions, with endografts that are able to be surveilled postoperatively with ultrasound examination. None. None.
Clinical case
clinical
en
0.999996
PMC11697044
A 68-year-old man was referred to the vascular surgery department for evaluation of an EIA aneurysm incidentally found on a screening magnetic resonance imaging after an elevated prostate-specific antigen on routine screening laboratory tests. Medical history was significant for type 2 diabetes mellitus, hypertension, hyperlipidemia, and coronary artery disease without a history of myocardial infarction or preventative intervention. He presented to the office and was evaluated for symptoms; he reported none. There was no history of trauma, infectious etiology, or prior vascular access. The patient denied any history of smoking, cycling or extreme sporting, or family history of aneurysms. Blood cultures were negative, and leukocytes were within normal limits. Computed tomography angiogram of the chest, abdomen, and pelvis with runoff revealed an isolated right sided saccular 2.6-cm EIA aneurysm above the inguinal ligament, with no extension proximally or distally . There was no aneurysmal or major atherosclerotic disease in the abdominal aorta or distal arterial vessels and there were no signs of disease in the contralateral iliac arteries. Based on discussion with the vascular surgery team, the patient was given the options of endovascular vs open surgery. Using shared decision-making, an endovascular approach was chosen to treat the isolated EIA aneurysm, and the patient provided consent. Fig 1 Preoperative computed tomography angiography showing an isolated aneurysm of the suprainguinal right external iliac artery ( EIA ). ( A ) Sagittal. ( B ) Axial. ( C ) Coronal with measurement. The operation was performed with the patient under general anesthesia. An 8F short 25 cm sheath (Terumo Medical Co, Tokyo, Japan) was placed percutaneously at the left femoral artery, and the Omni Flush Soft-Vu Angiographic Catheter (Angiodynamics, Latham, NY) was advanced and positioned into the infrarenal abdominal aorta. An aortogram was captured to visualize the iliac arteries with an oblique view to visualize the right hypogastric takeoff. A widely patent bilateral iliac system was visualized, and a large 2.6-m aneurysm was identified 4 cm above the femoral bifurcation. The Omni Flush over a floppy Glidewire (Terumo Medical Corp., Somerset, NJ) was advanced across the iliac bifurcation, beyond the aneurysm sac, and further down into the right superficial femoral artery. The short 8F sheath was exchanged for an 8F 45 cm Ansel Sheath (Cook Medical, Bloomington, IN) over a J-tipped Stiff Amplatz Wire (Boston Scientific, Natick, MA), and advanced to the mid-right EIA just proximal to the aneurysm. After heparinizing the patient and measuring the native vessel for optimal graft selection, the 9 mm × 10 cm Viabahn stent graft (W. L. Gore & Associates, Flagstaff, AZ) was ultimately selected and carefully deployed in a distal to proximal fashion. The entirety of the aneurysmal sac was covered while ensuring maintaining patency of the common femoral artery distally and hypogastric artery proximally. Final angiography demonstrated widely patent right EIA and widely patent femoral bifurcation with complete exclusion of the EIA aneurysm. Given that the stent graft appeared to have an excellent seal, it was decided not to post-dilate with balloon angioplasty. Fig 2 Intraoperative angiography. ( A ) Preoperative aortogram. ( B ) Preoperative iliac angiogram. ( C ) Completion angiogram after stent graft deployment. ( D ) Reconstructed three-dimensional image of 2-week follow-up computed tomography scan showing completely excluded aneurysm with patent stent graft in suprainguinal right external iliac artery ( EIA ). The patient was discharged home on postoperative day 1 without complication on a regimen of aspirin only, indefinitely. On outpatient review at 2 weeks, the patient was well. No difference in peripheral pulse examination was found. The patient was asymptomatic preoperatively and remained symptom free at the 2-week follow-up. Postoperative computed tomography angiography demonstrated an excluded aneurysm sac with a good apposition of the stent graft with no evidence of endoleaks or stent graft-related complications . We plan to perform annual ultrasound surveillance of the stent graft. Common and internal iliac artery aneurysms have multifactorial pathogeneses that are nearly identical to that of abdominal aortic aneurysms, as seen by their histological similarities. The particular rarity of aneurysms involving the EIA can be attributed to the unique lamellar architecture and biomechanical properties of the external iliac arterial walls, particularly in the tunica media. 7 Distinct from the more proximal aortoiliac segments, external iliac arteries possess a more structured and layered lamellar architecture, as well as a higher elastin-to-collagen ratio, 8 which allows them to withstand higher hemodynamic stresses and accommodate higher pressures, thus reducing the susceptibility to wall weakening and aneurysmal dilation. Isolated iliac artery aneurysms, without any other identifiable aortoiliac or peripheral vascular disease, have been described in multiple investigations to be a rare pathology. Silver et al 9 in 1967 performed a chart review of patients with arterial aneurysms affecting the aortic or iliac artery systems and found 571 patients with abdominal aortic aneurysms and only 11 patients with isolated iliac artery aneurysms, a relative frequency of 1.9%. Later in 1983, McCready et al 10 reported the frequency of isolated iliac artery aneurysms of 0.9% and provided one of the only anatomical frequency distributions amongst isolated aneurysms of the iliac artery system: 90% of all isolated iliac artery aneurysms affect the common iliac artery solely, whereas <1% affect the EIA solely. Finally, in 1989 Brunkwall et al 5 performed the largest investigation on isolated iliac artery aneurysms, reporting 13 cases during the 15-year compilation of autopsy and operating records in Malmo Sweden, population 230,000. They found only one isolated EIA aneurysm in that same study. 5 Regardless of how rare they are, many investigations have demonstrated that isolated iliac artery aneurysm are associated with a high risk of rupture and mortality, with rates of rupture between 14% and 75%. 2 , 11 , 12 The high mortality rate is postulated to be due to the lack of inclusion of iliac artery aneurysms in a differential diagnosis of pelvic conditions, partly owing to their rarity. Also, owing to the nature and location of the pathology, they are difficult to detect on physical exam until they are at a size when they are at risk for a morbid rupture. 3 McCready et al described that 78% of the patients in their study presented asymptomatically with their iliac artery aneurysm, similar to the patient described in this case study. Few cases have been reported describing isolated aneurysms of the EIA ( Table ). The first case report in 1952 described a patient who presented symptomatically with abdominal and lower limb pain. Surgical exploration revealed an EIA aneurysm that had ruptured. 13 The next three case reports published between 1986 and 2009 described iliac artery aneurysms involving the EIA that were found histologically to be due to cystic medial necrosis. 14 , 15 , 16 More recently, in 2019 two cases were presented with a 65-year-old symptomatic patient with left lower limb edema and a 55-year-old symptomatic patient with intermittent left thigh pain associated with paresthesias, both ipsilateral to the isolated EIA aneurysm. 17 , 18 Finally in 2020, there was another case report, similar to Crivello's, describing a symptomatic isolated EIA aneurysm associated with cystic medial necrosis. 19 In the present report, we have presented the case of a 68-year-old man who was completely asymptomatic, with an incidental finding of an EIA aneurysm. Until the presentation of our case, a search of PubMed found only seven reported cases of isolated EIA aneurysm, none of which were completely asymptomatic or repaired endovascularly. Table Reported cases of isolated external iliac artery ( EIA ) aneurysm Author Age, years Sex Size, cm Year Priddle et al 13 29 Female 4.0 1952 Crivello et al 14 27 Male Not available 1986 Mohan et al 15 66 Male 11.0 1997 Kato et al 16 78 Female 4.0 2009 Van de Luijtgaarden et al 17 65 Male 3.5 2019 Hussain et al 18 55 Male 7.0 2019 Chatzantonis et al 19 51 Male 2.0 2020 Current case 68 Male 2.6 2023 With this case report, we hope to stimulate a discussion on when to intervene on these rare pathologies. Although we have guidelines, based on large sample studies, for when to fix common or internal iliac artery aneurysms, the rarity of isolated EIA aneurysms leaves vascular surgeons without clear directions on when to fix them, especially when they are asymptomatic. Of the different publications reporting sizes of isolated EIA aneurysms, we found that most were repaired between 2 and 4 cm, mostly in men approximately 60 years old. Until clearer, large sample studies are performed, we recommend early repair of these aneurysms (diameter ≥2 cm) owing to the risk of rupture or symptoms seen in prior publications. Additionally, we now have the availability of minimally invasive interventions, with endografts that are able to be surveilled postoperatively with ultrasound examination. None. None.
Clinical case
clinical
en
0.999996
PMC11697044
A 68-year-old man was referred to the vascular surgery department for evaluation of an EIA aneurysm incidentally found on a screening magnetic resonance imaging after an elevated prostate-specific antigen on routine screening laboratory tests. Medical history was significant for type 2 diabetes mellitus, hypertension, hyperlipidemia, and coronary artery disease without a history of myocardial infarction or preventative intervention. He presented to the office and was evaluated for symptoms; he reported none. There was no history of trauma, infectious etiology, or prior vascular access. The patient denied any history of smoking, cycling or extreme sporting, or family history of aneurysms. Blood cultures were negative, and leukocytes were within normal limits. Computed tomography angiogram of the chest, abdomen, and pelvis with runoff revealed an isolated right sided saccular 2.6-cm EIA aneurysm above the inguinal ligament, with no extension proximally or distally . There was no aneurysmal or major atherosclerotic disease in the abdominal aorta or distal arterial vessels and there were no signs of disease in the contralateral iliac arteries. Based on discussion with the vascular surgery team, the patient was given the options of endovascular vs open surgery. Using shared decision-making, an endovascular approach was chosen to treat the isolated EIA aneurysm, and the patient provided consent. Fig 1 Preoperative computed tomography angiography showing an isolated aneurysm of the suprainguinal right external iliac artery ( EIA ). ( A ) Sagittal. ( B ) Axial. ( C ) Coronal with measurement. The operation was performed with the patient under general anesthesia. An 8F short 25 cm sheath (Terumo Medical Co, Tokyo, Japan) was placed percutaneously at the left femoral artery, and the Omni Flush Soft-Vu Angiographic Catheter (Angiodynamics, Latham, NY) was advanced and positioned into the infrarenal abdominal aorta. An aortogram was captured to visualize the iliac arteries with an oblique view to visualize the right hypogastric takeoff. A widely patent bilateral iliac system was visualized, and a large 2.6-m aneurysm was identified 4 cm above the femoral bifurcation. The Omni Flush over a floppy Glidewire (Terumo Medical Corp., Somerset, NJ) was advanced across the iliac bifurcation, beyond the aneurysm sac, and further down into the right superficial femoral artery. The short 8F sheath was exchanged for an 8F 45 cm Ansel Sheath (Cook Medical, Bloomington, IN) over a J-tipped Stiff Amplatz Wire (Boston Scientific, Natick, MA), and advanced to the mid-right EIA just proximal to the aneurysm. After heparinizing the patient and measuring the native vessel for optimal graft selection, the 9 mm × 10 cm Viabahn stent graft (W. L. Gore & Associates, Flagstaff, AZ) was ultimately selected and carefully deployed in a distal to proximal fashion. The entirety of the aneurysmal sac was covered while ensuring maintaining patency of the common femoral artery distally and hypogastric artery proximally. Final angiography demonstrated widely patent right EIA and widely patent femoral bifurcation with complete exclusion of the EIA aneurysm. Given that the stent graft appeared to have an excellent seal, it was decided not to post-dilate with balloon angioplasty. Fig 2 Intraoperative angiography. ( A ) Preoperative aortogram. ( B ) Preoperative iliac angiogram. ( C ) Completion angiogram after stent graft deployment. ( D ) Reconstructed three-dimensional image of 2-week follow-up computed tomography scan showing completely excluded aneurysm with patent stent graft in suprainguinal right external iliac artery ( EIA ). The patient was discharged home on postoperative day 1 without complication on a regimen of aspirin only, indefinitely. On outpatient review at 2 weeks, the patient was well. No difference in peripheral pulse examination was found. The patient was asymptomatic preoperatively and remained symptom free at the 2-week follow-up. Postoperative computed tomography angiography demonstrated an excluded aneurysm sac with a good apposition of the stent graft with no evidence of endoleaks or stent graft-related complications . We plan to perform annual ultrasound surveillance of the stent graft. Common and internal iliac artery aneurysms have multifactorial pathogeneses that are nearly identical to that of abdominal aortic aneurysms, as seen by their histological similarities. The particular rarity of aneurysms involving the EIA can be attributed to the unique lamellar architecture and biomechanical properties of the external iliac arterial walls, particularly in the tunica media. 7 Distinct from the more proximal aortoiliac segments, external iliac arteries possess a more structured and layered lamellar architecture, as well as a higher elastin-to-collagen ratio, 8 which allows them to withstand higher hemodynamic stresses and accommodate higher pressures, thus reducing the susceptibility to wall weakening and aneurysmal dilation. Isolated iliac artery aneurysms, without any other identifiable aortoiliac or peripheral vascular disease, have been described in multiple investigations to be a rare pathology. Silver et al 9 in 1967 performed a chart review of patients with arterial aneurysms affecting the aortic or iliac artery systems and found 571 patients with abdominal aortic aneurysms and only 11 patients with isolated iliac artery aneurysms, a relative frequency of 1.9%. Later in 1983, McCready et al 10 reported the frequency of isolated iliac artery aneurysms of 0.9% and provided one of the only anatomical frequency distributions amongst isolated aneurysms of the iliac artery system: 90% of all isolated iliac artery aneurysms affect the common iliac artery solely, whereas <1% affect the EIA solely. Finally, in 1989 Brunkwall et al 5 performed the largest investigation on isolated iliac artery aneurysms, reporting 13 cases during the 15-year compilation of autopsy and operating records in Malmo Sweden, population 230,000. They found only one isolated EIA aneurysm in that same study. 5 Regardless of how rare they are, many investigations have demonstrated that isolated iliac artery aneurysm are associated with a high risk of rupture and mortality, with rates of rupture between 14% and 75%. 2 , 11 , 12 The high mortality rate is postulated to be due to the lack of inclusion of iliac artery aneurysms in a differential diagnosis of pelvic conditions, partly owing to their rarity. Also, owing to the nature and location of the pathology, they are difficult to detect on physical exam until they are at a size when they are at risk for a morbid rupture. 3 McCready et al described that 78% of the patients in their study presented asymptomatically with their iliac artery aneurysm, similar to the patient described in this case study. Few cases have been reported describing isolated aneurysms of the EIA ( Table ). The first case report in 1952 described a patient who presented symptomatically with abdominal and lower limb pain. Surgical exploration revealed an EIA aneurysm that had ruptured. 13 The next three case reports published between 1986 and 2009 described iliac artery aneurysms involving the EIA that were found histologically to be due to cystic medial necrosis. 14 , 15 , 16 More recently, in 2019 two cases were presented with a 65-year-old symptomatic patient with left lower limb edema and a 55-year-old symptomatic patient with intermittent left thigh pain associated with paresthesias, both ipsilateral to the isolated EIA aneurysm. 17 , 18 Finally in 2020, there was another case report, similar to Crivello's, describing a symptomatic isolated EIA aneurysm associated with cystic medial necrosis. 19 In the present report, we have presented the case of a 68-year-old man who was completely asymptomatic, with an incidental finding of an EIA aneurysm. Until the presentation of our case, a search of PubMed found only seven reported cases of isolated EIA aneurysm, none of which were completely asymptomatic or repaired endovascularly. Table Reported cases of isolated external iliac artery ( EIA ) aneurysm Author Age, years Sex Size, cm Year Priddle et al 13 29 Female 4.0 1952 Crivello et al 14 27 Male Not available 1986 Mohan et al 15 66 Male 11.0 1997 Kato et al 16 78 Female 4.0 2009 Van de Luijtgaarden et al 17 65 Male 3.5 2019 Hussain et al 18 55 Male 7.0 2019 Chatzantonis et al 19 51 Male 2.0 2020 Current case 68 Male 2.6 2023 With this case report, we hope to stimulate a discussion on when to intervene on these rare pathologies. Although we have guidelines, based on large sample studies, for when to fix common or internal iliac artery aneurysms, the rarity of isolated EIA aneurysms leaves vascular surgeons without clear directions on when to fix them, especially when they are asymptomatic. Of the different publications reporting sizes of isolated EIA aneurysms, we found that most were repaired between 2 and 4 cm, mostly in men approximately 60 years old. Until clearer, large sample studies are performed, we recommend early repair of these aneurysms (diameter ≥2 cm) owing to the risk of rupture or symptoms seen in prior publications. Additionally, we now have the availability of minimally invasive interventions, with endografts that are able to be surveilled postoperatively with ultrasound examination. None. None.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11697514
Central pancreatectomy has emerged as an effective therapeutic alternative for the management of benign and low-grade pancreatic tumors, especially in cases where the preservation of pancreatic function is crucial, and minimizing morbidity associated with more radical resections, such as distal pancreatectomy or pancreatoduodenectomy, is desired . Unlike these traditional techniques, central pancreatectomy allows for the resection of localized neoplasms without significantly compromising the surrounding pancreatic tissue, resulting in a reduced risk of postoperative pancreatic insufficiency . Recent studies support central pancreatectomy as a valid therapeutic option in selected clinical contexts, where multidisciplinary preoperative evaluation plays a fundamental role in case selection. This article presents a clinical case of central pancreatectomy to contribute to the understanding of its role as a therapeutic option in the treatment of low-grade pancreatic tumors and its impact on pancreatic tissue preservation. A 45-year-old female patient with a history of conversion from sleeve gastrectomy to Roux-en-Y gastric bypass three years ago due to gastroesophageal reflux disease (GERD) presented with a pancreatic cystic lesion found incidentally on abdominal ultrasound screening . Further investigation with magnetic resonance imaging (MRI) revealed a 20 mm cystic lesion in the neck of the pancreas without features of malignancy . Endosonography showed no findings suggestive of aggressiveness. Pancreatic fine-needle aspiration (FNA) was performed (Table 1 ), with citrine-colored fluid aspirated and carcinoembryonic antigen (CEA) < 1.8 ng/mL. The biochemical analysis of the fluid showed amylase of 144 U/L; glucose of 102 mg/dL; immunohistochemistry, chromogranin A diffusely positive in neoplastic cells; synaptophysin, diffusely positive in neoplastic cells; and Ki-67, proliferation index estimated at <1%, consistent with well-differentiated grade 1 neuroendocrine tumor (G1 NET). Preoperative laboratory tests were performed, showing serum chromogranin A within the normal reference range (Table 2 ). PET-CT with octreotide showed uptake in the pancreas with no other lesions. Given the suspicion of a neuroendocrine pancreatic neoplasm, the case was discussed in a clinical committee, and an open central pancreatectomy was decided. The surgery was performed through a midline laparotomy, with the opening and section of the gastrocolic ligament, providing access to the lesser sac and full exposure of the pancreas. Macroscopically, a soft pancreas with a well-defined, partially exophytic cystic lesion in the neck, approximately 20 mm in its largest diameter, was identified, involving almost the entire thickness of the pancreatic parenchyma . The lymph node dissection of group 8 was performed, along with the dissection of the common hepatic artery and splenic artery. The dissection of the pancreatic groove to the left of the mesenteric vessels allowed the creation of a retro-pancreatic tunnel without complications. To the left, the splenic vein was identified in its usual position. The tunnel was completed using blunt dissection and an esophageal retractor to encircle the pancreatic body, achieving wide proximal and distal margins . A macroscopic view revealed a well-defined, partially exophytic cystic neoplasm located in the neck of the pancreas. The pancreatic neck was fully exposed, ensuring wide distal and proximal margins. The transection of the pancreatic neck was performed using an Endo GIA (Covidien, Dublin, Ireland) 60 mm purple cartridge, and distal pancreas resection was completed with monopolar energy . The Wirsung duct was identified with a diameter of approximately 2-3 mm . The jejunum was transected 20 cm distal to the previous entero-entero anastomosis of the gastric bypass. A transmesocolic loop was brought up, and a Blumgart pancreatojejunostomy was created with 10 separate duct-to-mucosa Prolene 5-0 stitches . A side-to-side mechanical entero-entero anastomosis was performed. Two Blake drains were placed in the pancreatic bed, with distal ends adjacent to the pancreatic stump and exteriorized through the right flank. On postoperative day 4, both drains had minimal output (drain I, 10 cc; drain II, 59 cc) with amylase levels of 5878 and 59 mg/dL, respectively (Table 3 ). Drain II was removed, and the patient was discharged on postoperative day 5 due to favorable clinical progress. She was evaluated eight days post-discharge; drain I output was 3-5 cc/day, leading to its removal. Biopsy confirmed a well-differentiated 2.1 cm G1 neuroendocrine tumor. Surgical margins were negative, with no vascular, lymphatic, or perineural invasion (pT2N0). At the five-month follow-up, the patient was asymptomatic, with a control abdominal PET-CT showing no abnormalities. Traditional approaches, such as distal pancreatectomy and pancreatoduodenectomy, involve more extensive resections, which may result in a higher risk of postoperative complications, including diabetes and malabsorption . Authors such as Iacono et al. support the idea that central pancreatectomy may be superior to distal pancreatectomy in certain contexts, particularly for patients with benign or low-grade tumors. Central pancreatectomy offers greater pancreatic tissue preservation and a lower rate of severe complications compared to distal pancreatectomy, making it the preferred surgical option in selected cases . The choice of surgical technique should consider not only the type of tumor but also the patient's clinical characteristics and the surgical context. Multidisciplinary clinical evaluation is essential for decision-making, where surgical planning plays a fundamental role. In terms of pancreatic function preservation, a recent study by Lee et al. directly compared central pancreatectomy, distal pancreatectomy, and duodenopancreatectomy. Central pancreatectomy demonstrated significant advantages in terms of pancreatic functional preservation, with favorable long-term outcomes not only for pancreatic function but also in preserving pancreatic mass, which ultimately translates into a lower incidence of postoperative diabetes and an improved quality of life for patients . Regarding the effectiveness and safety of central pancreatectomy, a recent systematic review and meta-analysis established its feasibility for both open and minimally invasive techniques. Although minimally invasive techniques offer additional benefits in terms of reducing surgical trauma, recovery time, and hospital stay, central pancreatectomy remains effective in open surgery . The adoption of minimally invasive techniques developed in recent years represents a current challenge, with laparoscopic approaches being a safe technique and an important advancement in surgical practice . Despite the numerous advantages of central pancreatectomy, the technique requires a high level of skill and experience from the surgeon, which may limit its application in centers with less experience in pancreatic surgery. In our case, the surgical team had extensive experience in pancreatic surgery, enabling central pancreatectomy to be considered as a therapeutic option, with thorough preoperative evaluation and planning. We believe that continuous training in minimally invasive techniques and the establishment of standardized protocols play a fundamental role in improving surgical outcomes in these cases. The reviewed studies support central pancreatectomy as a valid therapeutic option and, in certain cases, a preferred choice, particularly with the current advancements in minimally invasive techniques.
Clinical case
clinical
en
0.999996
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699598
Out-of-hospital-cardiac arrest (OHCA) is a significant health concern with approximately 275,000 annual cases in Europe and 420,000 in the United States. 1 Immediate recognition of cardiac arrest, initiation of cardiopulmonary resuscitation (CPR) and early defibrillation are key factors to improve survival. 2 However, bystander CPR are only reported on average in 58 % (range 13 % to 83 %) of episodes. 2 In addition, there is considerable variation in the quality of bystander CPR, with only a minority demonstrating high quality CPR. 3 Simulation studies have shown that video assisted CPR (V-CPR) may improve cardiac arrest recognition, improve the quality of basic CPR and that it can be performed in a real-life setting. 4 , 5 In OHCA, clinical studies show promising results of V-CPR on CPR quality, return-of-spontaneous-circulation (ROSC) rates and survival to hospital discharge. 6 , 7 , 8 , 9 However, there are considerable knowledge gaps regarding possible benefits of applying this technology in patients with cardiac arrest. 10 We present a case with witnessed OHCA, where dispatcher V-CPR was initiated early and considered essential to provide a tailored approach to a complex OHCA. A 58-year-old male with known paroxysmal atrial fibrillation (AF) sustained an OHCA at 9:47 a.m. while changing tires on his jacked-up car together with his son. An emergency call was received at 9:48 a.m. The patient was reported unconscious with agonal respiration and no CPR was performed. Guided by the Norwegian Index for Medical Emergency Assistance, a stepwise tool for decision-making and to guide callers in emergency medical situations by the Emergency Medical Coordination Centre (EMCC), the patient was identified as unresponsive and not breathing normally. 11 The medical dispatcher immediately instructed the two laypersons on-scene to perform CPR, which they did without hesitation. None had previous CPR experience. Emergency medical services (EMS) and the on-call general practitioner were also immediately dispatched. No other emergency resources (e.g. volunteer first responders) were available during the incident. Since there were two persons on-scene, the medical dispatcher decided to initiate a video call (VC) to better guide the ongoing layperson CPR. The video transmission was initiated two minutes after the emergency call and showed high-quality chest compressions, and a patient with apparent spontaneous respiration and limb movements. After three minutes, they paused compressions according to Norwegian CPR guidelines. 12 The medical dispatcher then observed that the patient‘s breathing pattern changed into agonal respiration. When compressions were continued, the respiration was again considered normal. Because the breathing pattern was deemed normal due to well performed chest compressions, the medical dispatcher decided not to instruct in mouth-to-mouth ventilations according to standard CPR guidelines. The patient therefore received compression-only CPR until arrival of EMS at 10.21 am, 33 min after start of bystander CPR, at which point V-CPR was discontinued. The two laypersons alternated in performing CPR during the whole incident prior to EMS arrival. On EMS arrival, the patient was still in cardiac arrest and presented with ventricular fibrillation (VF). A direct current (DC) defibrillation with 200 Joule was delivered by EMS personnel before they continued CPR. The patient obtained ROSC at 10:23 am. However, the ROSC only lasted for 30–40 s, and the patient resumed to VF. Another two DC 200 J defibrillations were delivered, but the patient remained with VF. A physician-staffed air ambulance helicopter arrived on-scene at 10:30 am. With ongoing CPR with the patient in VF, the patient showed signs of life with head and limb movements, verbal sounds and a normal respiration pattern with a respiration rate of 12 breaths per minute during chest-compressions. A fourth DC 200 Joule defibrillation was delivered which resulted in sustained ROSC with AF rhythm at 10:33, 46 min after the time of cardiac arrest. Before transportation a rapid sequence induction with intravenous (iv) analgesia (fentanyl 0.2 mg iv), sedation (ketamine 75 mg iv), muscle relaxation (rocuronium 50 mg iv) and endotracheal intubation was performed without any complications. An arterial line was inserted and an intravenous ketamine infusion (1.5 mg/kg/hour) was started for maintenance of sedation during transport to hospital. The patient was hemodynamically stable in-flight with a blood pressure of 150/80 and a pulse rate of 70–90/min, except a brief period of ventricular tachycardia during take-off from the scene. A dose of intravenous amiodarone 100 mg was administered, and the patient converted to AF. The measured oxygen saturation level was 98 % and the patient was normothermic. Transport to the nearest university hospital was uneventful and he was admitted to hospital at 11:59 a.m. The first arterial blood gas showed a metabolic acidosis with pH 7.20, pCO2 5.7 kPa, pO2 12.2 kPa, base excess −11.2 mmol/L, lactate 5.5 mmol/L, bicarbonate 17 mmol/L and glucose 13.6 mmol/L. Other values were normal. A percutaneous coronary intervention was performed with a successful revascularisation and stent implementation of partly occluded left anterior descending and circumflex arteries. A one-chamber cardioverter-defibrillator was implanted and he was discharged to a local hospital for follow-up on day 11, with full neurological recovery. Access to timely EMS response in time-critical situations (e.g. cardiac arrest) is a vital factor to improve patient outcome. 13 The EMS response time in this case was 33 min, which is often the case in sparsely populated areas, such as rural Norway. 14 However, the massive development of smartphone technology during the last two decades has opened new telemedicine possibilities in modern healthcare. 15 The use of VC to assist callers and EMS has the potential to compensate for longer response and travel times. However, despite considerable interest in this technological development, the evidence of effect on patient survival and outcome in real-life OHCA is limited as the specific effect of V-CPR has been difficult to isolate. Other promising technological developments, such as the use of drones carrying defibrillators in OHCA, have shown the potential for increased access to advanced devices and additional decision-making, especially in rural areas. 16 , 17 However, like V-CPR, there is a need for further testing in real-life situations to evaluate efficiency and how these interventions impact final outcome. 16 , 17 A recent meta -analysis found that V-CPR can improve bystander CPR during simulated cases, but that the process is substantially affected by poor video signals and lack of guidance procedures. 18 Several studies have found improvements in specific items of the CPR process, with better compression rates and hand placement. 10 , 5 , 6 , 7 In our opinion, the main effect of V-CPR in this case was a more tailored approach and thorough guidance of laypersons without previous CPR experience in a complex setting with long-lasting CPR without back-up of present EMS, resulting in high-quality CPR, CPR induced consciousness (CPRIC) and a good neurological outcome. VCs were introduced in 2020 in Norwegian EMCCs for the use at the medical dispatcher’s discretion, in order to increase dispatcher situational awareness and improve the quality of care given by lay persons. 19 Currently, no specific guidelines exist on how VC can be used in the most appropriate way. The solution used in this case was developed by the Norwegian Air Ambulance Foundation and implemented in EMCC nationally in collaboration with the Norwegian Directorate of Health in 2020. 20 The EMCC sends a link to the caller’s mobile phone after approval by the caller, and the video link allows the medical dispatcher to use the available mobile phone camera. 20 , 21 Preliminary evaluations pertaining to general use have described high user-friendliness and that the medical dispatcher’s perception of patient’s acuity was affected in about half of the cases. 19 We strongly believe that video assistance by trained medical personnel has future unsolved potential. However, it is important to recognize the need for 1) clear guidance protocols 2) training in video-assisted dispatch and 3) training in the ability to instruct lay rescuers in psychological distress. It is also important to keep in mind, that the availability of live video transmission from scenes may expose the EMCC dispatchers to unpleasant visual impressions. 22 . Apparent signs of life (i.e. breathing and head/limb movements) were observed in this case during compressions by the laypersons and after arrival of EMS personnel. This situation added extra complexity, which potentially could have affected the decision to continue or stop ongoing CPR. In case reports and a systematic review describing the phenomena, the observation of breathing efforts has been inconsistently reported. 23 , 24 Agonal gasps or breathing efforts also may be present in patients with cardiac arrest not receiving CPR, so this sign may be less sensitive on CPRIC than movements. Increasing number of CPRIC events are reported, most likely due to a combined effect of community CPR responder programs, improved CPR quality and increased focus on the chain of survival. 25 A noteworthy point is that in a prospective study focusing on cognitive experiences in cardiac arrest survivors, only 2 % of patients retrieved visual or auditory awareness following their incident and no patients remembered any experience of pain. 26 In the described case, the patient could not recall any memory of the incident. The case represents a complex cardiac arrest with long-lasting CPR, signs of life and no previous CPR experience in lay persons, where V-CPR was instrumental in providing on-scene guidance and decision-making. Steinar Einvik: Writing – review & editing, Writing – original draft, Investigation, Data curation, Conceptualization. Ole Erik Ulvin: Writing – review & editing. Trond Nordseth: Writing – review & editing. Oddvar Uleberg: Writing – review & editing, Writing – original draft, Supervision, Investigation, Data curation, Conceptualization. The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
en
0.999997
PMC11699881
Carotid cavernous fistulas are a serious condition that involves spontaneous or trauma-induced disruption in the carotid arteries within the cavernous sinus. This disruption causes blood to leak into the sinus, which impedes the function of cranial nerves III, IV, V1, V2, and VI . Early signs of CCF often include proptosis, glaucoma, severe headaches, partial blindness, and conjunctival hyperemia, especially in patients with preexisting conditions such as atherosclerosis, hypertension, and diabetes. If left untreated, patients can develop periorbital disfigurement and permanent vision loss . Despite effective methods of diagnosis using angiography and blood vessel repair, this condition is often misdiagnosed due to it making up only 1.5% of endovascular-treated ruptured aneurysms and its similarity in presentation to cerebrospinal fluid (CSF) leaks . Therefore, CCFs have been under-investigated, and additional research is required to delineate the cause, unique features, and proper treatment of this phenomenon. Often misdiagnosed due to its ambiguous presentation, we present a successful clinical workup and treatment of a 78-year-old female patient who developed a CCF in the setting of spinal fusion. We suggest a potential relationship between prolonged prone positioning during surgical intervention and the occurrence of CCF. A 78-year-old female patient presented for bilateral lateral radicular leg discomfort and back pain refractory to conservative management including physical therapy and anti-inflammatory medications. Magnetic resonance imaging (MRI) of the lumbar spine revealed significant degenerative spondylosis of L4-S1 levels including severe lateral recess stenosis at L4-L5 and bilateral pars defects at L5-S1 . She was neurologically intact with no remarkable findings on physical exam. Operative management was pursued due to her symptoms and imaging findings. She underwent a successful L4-S1 posterior instrumented fusion with partial laminectomies at L4 and L5 along with an L4-L5 bilateral facetectomy. There was no significant intraoperative fluid leak. Estimated blood loss was minimal, and the procedure lasted approximately 228 minutes with the patient prone on a Jackson spine table. The patient’s postoperative course was uneventful until postoperative day 3 when the patient developed severe throbbing headaches and neck and shoulder pain/stiffness that resolved with laying flat. The patient’s symptoms resolved the following day, so she was discharged. Two weeks following her surgery, she presented for suture removal and had no complaints of headaches or neck and shoulder pain. Six weeks following the surgery, the patient presented for a follow-up with complaints of bifrontal headaches that were worse in the morning for the past two weeks. She also reported neck discomfort and sharp discomfort whenever she turned her head to one side or extended her neck. The headaches were not positional nor particularly severe. There was no evidence of a spinal fluid leak from the incision nor palpable fluid collection appreciated on the physical exam. An x-ray of the cervical spine demonstrated degenerative changes from C4 through C7 with complete loss in disc space height at each level from C4 inferiorly . The patient presented to the Emergency Department on two separate occasions in the following two weeks; first, she had a pounding headache in bilateral temples with swelling below her left eye and an episode of emesis the evening prior. She was also noted to have conjunctivitis in both eyes and was given antibiotic eye drops. She presented the second time with complaints of a constant headache in the middle of the night, nausea, and new onset blurry vision in her left eye. The patient was also hypertensive with 170 systolic blood pressure. A brain CT scan showed no evidence of an acute intracranial process. Basic lab workup was unremarkable on both visits. The patient was discharged the second time with prescriptions for Percocet and Zofran and was recommended to follow up with her neurologist. Eight weeks following her fusion, the patient was struggling with worsening headaches and nausea. A lumbar spine MRI showed a fluid collection adjacent to the dura with a possible dural tear . Her worsening symptoms prompted a return to the operating room for wound exploration and repair of a suspected CSF leak. There was no apparent CSF leak. Valsalva was performed with no obvious egress of spinal fluid. Given that there may have been an occult non-identifiable leak the dura was covered with two layers of Surgicel (Ethicon, Johnson & Johnson, USA) followed by Adherus (HyperBranch Medical Technology, Durham, NC, USA) tissue sealant. A lumbar drain was placed through the next most rostral intact interspinous space brought out through a separate stab incision. A 10-round Jackson-Pratt drain was tunneled into the sub-fascial space and brought out through an additional stab incision. Following the surgery, she had two episodes of emesis with continued headaches and nausea. However, she experienced significant improvement in her headaches and nausea over the subsequent days. She was discharged one week later following drain removal and complete resolution of her symptoms. Two weeks following discharge, she presented to the emergency department again for continued headaches, left conjunctival hemorrhage, mild proptosis, diplopia, and a cranial nerve (CN) III palsy. Given the unclear etiology for her headaches and new exam findings, there was a concern for Tolosa-Hunt syndrome or a carotid-cavernous fistula which prompted a brain MRI. Imaging demonstrated prominent superior ophthalmic veins, with the right being greater than the left . A left carotid-cavernous fistula was confirmed using diagnostic angiography. The patient underwent transvenous embolization for carotid-cavernous fistula in the intercavernous and left cavernous sinus. The fistula was nearly completely occluded with a significant reduction of flow into the fistula and gradual occlusion noted during angiography from the arterial supply . She had a successful coil embolization of a Barrow Type C carotid-cavernous fistula. Following the procedure, the patient had a significant improvement in her symptoms and was discharged three days later. Her postoperative course was significant for an improvement in her headaches and a lingering CN III and VI palsy predominantly of the left eye. Carotid-cavernous fistulas (CCF) are pathological shunts between the cavernous sinus and the carotid artery or its meningeal branches. The Barrow classification system anatomically describes four types of CCFs. Type A CCFs are direct, high-flow communications between the cavernous sinus and the internal carotid artery (ICA) as it traverses the sinus. Low-flow fistulas indirectly connect the carotid artery to the cavernous sinus via the ICA meningeal branch (Type B), external carotid artery meningeal branch (Type C), or both branches (Type D) . Several etiologies regarding the pathogenesis of CCF have been described. Type A CCFs are often associated with trauma, ruptured ICA aneurysms, connective tissue diseases, or iatrogenic injury . Type A CCF has been reported after various procedures, particularly those involving direct carotid manipulation or involvement of adjacent structures; procedures reported in association with Type A CCF include carotid endarterectomy, mechanical thrombectomy, transsphenoidal exploration, craniotomy and ocular surgery . Traumatic CCFs may occur as a result of bony fragment damage to the vessel wall secondary to basilar skull fracture or shear forces directly damaging the vessel wall . Indirect CCFs most commonly affect elderly women and are classically associated with ICA dissection, hypertension, and connective tissue diseases, including atherosclerosis, fibromuscular dysplasia, and Ehlers-Danlos syndrome . The presenting signs and symptoms of CCF are variable, depending on the characteristics of the CCF. Direct CCF often presents with conjunctival injection, blurred vision, headache, pulsating proptosis, and strabismus, most often affecting the sixth cranial nerve (the only cranial nerve traversing the interior of the cavernous sinus) . Low-flow CCFs often follow an insidious course, and conjunctival injection is typically the predominant physical complaint . Notably, low-flow CCFs are especially subject to misdiagnosis; our case represents a delayed diagnosis due to the patient’s recent spinal surgery and symptomatology mimicking a dural tear. The diagnosis of CCF is dependent upon imaging. Computed tomography (CT) and magnetic resonance (MR) imaging with or without angiography are often the first modalities utilized in assessing possible CCF and may be useful in diagnosing CCF; however, digital subtraction angiography (DSA) is currently considered the “gold standard” imaging modality for CCF because it demonstrates the cavernous sinus filling and drainage patterns . Strong clinical suspicion or CT/MRI evidence of possible CCF should prompt DSA . The exact treatment strategy is dependent upon the characteristics of the CCF. Direct CCFs are most often treated through endovascular approaches, with embolic materials, such as coils, acrylic glue, or ethylene vinyl alcohol copolymer, injected into the cavernous sinus to close fistula patency . Endoluminal stenting may be utilized to prevent subsequent embolism and promote endothelial proliferation . Twenty to sixty percent of indirect CCFs spontaneously resolve; thus, conservative management is usually attempted before intervention . External manual carotid compression with the contralateral hand several times per day has been shown to promote fistula closure . If surgical intervention is indicated, the same endovascular techniques used to repair direct CCFs are the first-line treatment modalities . However, because indirect CCFs involve smaller, tortuous carotid branches, endovascular therapy is not as effective compared to direct CCFs . Overall, endovascular treatment has a greater than 80% cure rate for CCF . If first-line treatments fail, open surgical ICA ligation can be attempted . Our patient was successfully treated with CCF coil embolization. CCFs are easily misdiagnosed, as CCF presentations can be highly variable and may be similar to several different diseases . Specific clinical histories may further confound the diagnosis of CCF. For example, Jain et al. describe a CCF that was initially misdiagnosed as hemorrhagic choroidal detachment following ocular surgery . Similarly, our patient's history of recent spinal surgery made a CSF leak a more plausible diagnosis than CCF. CSF leaks can present very similarly to CCF, with headaches, nausea, neck or back tightness, and cranial nerve palsies, being commonly shared symptoms . The diagnosis of CCF in our case was delayed because the patient’s recent surgical history made a dural tear more plausible. However, surgical exploration and drainage did not improve the patient’s symptoms, suggesting a dural tear was not present. After ruling out a dural tear, subsequent workup led to the diagnosis and treatment of CCF. To our knowledge, there is no association between CCF and spinal surgery or prolonged periods of prone positioning. There have been no reported cases of CCF following surgery not involving the carotid artery or related structures; all reported iatrogenic causes of CCF involve direct carotid involvement or surgery to adjacent structures . The patient has no known vascular diseases and suffered no recent trauma. Indirect CCFs (such as the Type C CCF seen in this patient) disproportionately affect elderly women . We hypothesize that perhaps a prolonged period spent in the prone position during her spinal surgery is responsible for the CCF in a patient whose demographics predisposed her to CCF. Our case represents the first reported CCF occurring in conjunction with spine surgery. Although unsubstantiated, we believe there may be an association between a prolonged period in the prone position and the development of CCF. Furthermore, we would like to document our clinical workup; in which the clinical history and symptomatology suggested a dural tear, although subsequent workup led to the diagnosis of CCF. This case demonstrates a difficult diagnosis of CCF and its potential relationship to spinal surgery.
Clinical case
clinical
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