pyomyoma ( suppurative leiomyoma ) is a rare , but life - threatening , condition resulting from infarction and infection of uterine leiomyoma ( 1,2 ) .
incidence of pyomyoma has decreased due to the development of antibiotics . since 1945 , only 50 pyomyoma cases have been documented in the literature , with a mortality rate of 6% ( 3/50 ) ( 35 ) . the most likely cause of mortality was delayed and difficult diagnosis . although the triad of pyomyoma are sepsis , leiomyoma , and no other source of infection ( 5 ) , it may present with silent or non - specific symptoms , which results in delayed diagnosis and treatment .
visualization of intratumoral gas formation may be suggestive of pyomyoma , but has not been consistently reported in all cases .
furthermore , large abdominal complex masses are likely to be first suspected as pelvic malignancies if found incidentally in perimenopausal women .
the present study experienced a rare case of large uterine pyomyoma in a perimenopausal woman who presented with anemia gravis , severe inflammatory reaction and cachexia .
a total of 50 reported cases of pyomyoma in the literature since 1945 were also studied .
a 53-year - old multigravida woman with 7 months of amenorrhea was referred to the department of obstetrics and gynecology ( wakayama medical university , wakayama , japan ) due to gradual abdominal distension starting 2 years previously .
the patient exhibited muscle weakness and walking difficulty , but no fever , abdominal pain or metrorrhagia .
her abdomen was swollen to the size of a beach ball , with a 126 cm abdominal circumference and body weight of 84.1 kg .
an intrauterine device ( iud ) had been inserted following her third birth , and no history of any lower abdominal or pelvic discomfort , leiomyoma , pelvic surgery , or other predisposing factors were known .
the present study was unable to determine the uterine cervix with pelvic examination due to its deviation , and was unable to acquire cytopathological findings of the cervix and endometrium .
transvaginal and transabdominal ultrasound examinations revealed a large abdominal mass with heterogeneous echogenicity . computed tomography ( ct ) and magnetic resonance imaging ( mri ) revealed a large unilocular mass with an irregular surface and thickened wall , occupying the abdominal cavity without gas formation ( fig .
1 ) . contrast ct revealed an expanded branch of the left internal iliac artery , which was suspected to be the uterine artery , surrounding the mass ( fig .
hemoglobin levels were 5.7 g / dl , white blood cell count was 57,300/l and c - reactive protein was elevated to 20.24 mg / dl .
cancer antigen ( ca)125 was also elevated to 200 u / ml . blood and vaginal cultures were negative .
possible diagnoses of the large abdominal mass included gynecologic tumor types ( benign or malignant ovarian tumor , uterine sarcoma or pyometra ) and gastrointestinal stromal tumor or mucocele of the appendix .
gastroscopy revealed no specific findings and colonoscopy revealed difficulty of insertion above the sigmoid colon due to pressure from the mass .
due to the finding of the uterine artery by contrast ct , the origin of the mass was suspected to be the uterus .
following antibiotic medication and blood transfusion , the patient underwent a total abdominal hysterectomy and bilateral salpingo - oophorectomy .
intraoperative findings demonstrated a solid tumor arising from the back of the uterine body with normal bilateral adnexa .
prior to removal , 12.4 liters of purulent , malodorous fluid was drained from the tumor ( fig .
the surrounding myometrium appeared normal and measured 3 cm in thickness , and the cut surface of the mass was purple - yellowish ( fig . 2c ) .
the pathological diagnosis was leiomyoma with marked necrosis and chronic inflammation , with no evidence of malignancy ( fig .
the post - operative course was uneventful and the patient was discharged from the hospital on post - operative day 14 . at 4 months follow - up , the patient 's weight had increased to 62 kg due to a good appetite .
pyomyoma occurs in both post - and pre - menopausal women , however , the risk of suppurative myoma is increased by pregnancy ( 1,2 ) . for post - menopausal patients , systemic vascular changes
necrosis of the leiomyoma caused by vascular flow insufficiency in the uterus following menopause is also a possible cause .
a history of uterine leiomyoma , pregnancy , abortion , menopause , uterine artery embolization ( uae ) , iud , vascular insufficiency ( diabetes , hypertension and atherosclerosis ) and systemic disease or infection may be predisposing factors for pyomyoma ; however , definitive diagnoses remain difficult .
although rapid clinical diagnosis for pyomyoma is often difficult due to its low incidence and the requirement to rule out the possibility of malignancy , mortality has decreased due to the improvement in surgical treatments , including myomectomy and hysterectomy , and broad - spectrum antibiotics .
a medline search since 1945 revealed only 50 reported cases of pyomyoma : 27 were non - pregnant woman ( mean age , 51.8 years ; range , 3669-years - old ; table i ) ( 329 ) and 23 cases were associated with pregnancy or abortion ( mean age , 33.6 years ; range , 2844-years - old ) .
the mean pyomyoma size in non - pregnancy and pregnancy - associated cases were 16.2 cm ( 338 cm ) and 13.5 cm ( 558 cm ) , respectively .
non - pregnancy pyomyoma tended to be larger compared with pregnancy - associated pyomyoma , and the present case was the largest among all reported cases of non - pregnancy - associated pyomyoma .
severe anemia gravis and inflammatory reaction were described in numerous pyomyoma cases , including the present study .
it has been shown that 2/50 cases had a history of iud usage ( 7,30 ) , 5/50 cases had uae ( 812 ) , 6/50 cases had vascular insufficiency ( 4,1317 ) and 8/50 cases demonstrated gas production ( 6,810,12,3133 ) .
although pyomyoma arises spontaneously , post - partum , post - instrumentation or post - surgery have been reported in the literature , only two cases include a iud , and only one case has been previously reported in a non - pregnant woman ( 7 ) .
infection by staphylococcus species was reported in 8 cases , streptococcus species in 7 cases , escherichia coli in 6 cases , enterococcus faecalis in 5 cases , clostridium species in 3 cases , proteus species in 2 cases and candida species in 2 cases . in the present case
, streptococcus agalactiae was cultured from the pus and tumor , however , not from the blood and vagina , suggesting the infection existed focally within the tumor .
therefore , conservative management with pre - operative broad - spectrum antibiotics and appropriate surgery was performed successfully . for surgical treatment ,
hysterectomy was performed in 32 cases , myomectomy in 10 cases and drainage in 6 cases .
diagnosis of large pyomyoma is difficult since surgery is required for a definitive diagnosis . a malignant tumor , in particular ovarian cancer ,
was initially suspected due to the findings of a large abdominal mass with signs of necrosis in a cachexic perimenopausal woman with an elevated ca125 level . in a previous report
, imaging analysis was shown to identify only non - specific results ( 6 ) .
although intratumoral gas formation is markedly suggestive of pyomyoma on ultrasound and ct imaging , gas formation is not consistently observed , as with the present case . in the present case , contrast ct contributed to the diagnosis of pyomyoma due to the identification of the uterine artery location .
ca125 has been observed to be increased in other gynecologic and non - gynecologic malignancies , as well as in a variety of benign disorders , including leiomyoma . among the previously reported cases of pyomyoma , 5 cases reported ca125 levels ( 6,7,14,15,18 ) , with a mean level of 56.3 u / ml ( 29.98109.7 u / ml ) .
ca125 was measured only in non - pregnancy - associated cases , possibly due to suspicion of gynecological malignancy based on the age of the patient and size of tumor .
furthermore , in pregnancy - associated cases , diagnosis of myoma was likely during pre - natal examination prior to the onset of the symptom , which may have contributed to the identification of the tumor origin . in the present case ,
to the best of our knowledge , this is the third iud - associated case of pyomyoma , with the largest tumor size and highest ca125 level compared with previously reported non - pregnancy cases .
a large pyomyoma is difficult to distinguish from a gynecological malignant tumor , particularly in perimenopausal , cachectic women with non - specific clinical presentation and without a history of leiomyoma .
a history of iud usage and contrast ct may be helpful in the diagnosis of pyomyoma .
gynecologists and general surgeons must be aware of the possibility of pyomyoma when presented with a large abdominal tumor , and in certain cases , surgery in combination with broad spectrum antibiotics may result in a good outcome .
 pyomyoma is a rare complication , which withoug antibiotics or surgical intervention , may cause sepsis and mortality . the present study reported a case of large uterine pyomyoma in a perimenopausal female . 
 a 53-year - old multigravida woman was referred to the department of obstetrics and gynecology ( wakayama medical university , wakayama , japan ) due to progressive abdominal distension . the patient presented with anemia gravis , severe inflammatory reaction and cachexia . 
 computed tomography revealed a large unilocular mass , 50 cm in size , with an irregular surface and thickened wall , occupying the entire abdomen . 
 following antibiotic medication , the patient underwent a total abdominal hysterectomy and bilateral salpingo - oophorectomy . 
 intraoperative findings demonstrated a solid tumor arising from the back of the uterine body . 
 a total of 12 liters of purulent , malodorous fluid was drained from the tumor . 
 the resected mass was 50 cm in size and 13.5 kg in weight . 
 cultures of the pus revealed the presence of streptococcus agalactiae . 
 pathological findings revealed suppurative leiomyoma with no malignancy . 
 large pyomyoma is difficult to distinguish from a gynecological malignant tumor types , particularly in perimenopausal women with non - specific clinical presentation . 
 although pyomyoma is a benign tumor , care must be taken to discriminate these from large abdominal tumors . 
Introduction
Case report
Discussion