A 40-year-old Indian gentleman with no known comorbid was referred to the otorhinolaryngology department for assessment of his nasal cavity and nasopharynx, following an incidental finding of a suspicious lesion within the left sphenoid sinus based on computerized tomography (CT). He presented with 2 weeks history of intermittent unilateral headache not relieved by conventional analgesics. It was described as throbbing in nature located at the left frontotemporal region. The pain was not precipitated by bending forward or coughing. He also did not complain of photophobia or autonomic symptoms. Unfortunately, his symptoms worsened with a new onset of double vision of 1-week duration. There was an absent history of fever, ear, nose, and throat symptoms. Further history did not suggest any possible risk factors for head and neck malignancy as well as immunodeficiency.\nClinically, he was well built with stable vital signs and a full Glasgow Coma Scale (GCS). The ophthalmological assessment demonstrated restrictions in all left extraocular muscle’s movement (Fig. ). The visual acuity of both eyes was not affected. However, relative afferent pupillary defect of his left eye was positive, evidenced by red desaturation of 50%. The anterior chamber, intraocular pressure, and fundi were unremarkable. In addition to that, the left corneal sensation was reduced, indicative of the fifth cranial nerve involvement. The rest of the cranial nerve’s functions were intact. Apart from oedematous mucosa overlying the left spheno-ehtmoidal recess, endoscopic evaluation of the nasal cavity revealed neither tumour growth nor purulent discharge from the paranasal sinus and nasopharynx. Assessment of the head and neck region was otherwise normal.\nCT unveiled calcified densities within areas of hypoattenuation in the left sphenoid sinus. No marked bony destruction was observed (Fig. ). A subsequent magnetic resonance imaging (MRI) was carried out to show hypodensity on T1 weighted post-contrast, and flow void in T2 weighted images within the same sinus (Figs. and ). Following informed consent, an emergency endoscopic left trans-ethmoidal sphenoidotomy was performed under general anaesthesia. A dense, darkened clay-like mass surrounded with the mucopurulent discharge was noticed within the left sphenoid sinus. The mucosa appeared inflamed and oedematous without clinical evidence of frank necrosis. The debris was utterly removed with a curette and sent for evaluation. The left sphenoid sinus was widened and thoroughly rinsed.\nHistopathological examination showed Candida species without evidence of mucosal invasion (Figs. and ) Intravenous ceftriaxone was commenced for a total duration of 14 days. His symptoms showed significant improvement 1 week after the operation with complete resolution of the headache. His left extraocular muscle functions were slowly restored at 4 weeks post-operation (Fig. ).
A 64-yr-old man presented to the emergency room with right visual loss after a 5-day history of rhinorrhea, diplopia and severe headache. The body temperature was 36.5℃ and the white blood cell count was 9,100/mm3. He had a 10-yr history of poorly controlled type II diabetes mellitus. The initial blood sugar was measured and it was too high to register by a blood glucose test strip and it was decreased to 485 mg/dL after an IV infusion of normal saline. His right eye showed ptosis, mild eyelid swelling and total ophthalmoplegia. His visual acuity in the right eye was counting fingers (50 cm) and it was 20/40 in the left eye. The pupillary light reflex was lost with an afferent defect. The anterior chamber, the intraocular pressure and the fundi were normal. Endoscopic examination detected edematous mucosa, brown-colored fungal balls and there was a mucopurulent discharge from the sphenoethmoidal recess. The opthalomologic and nasal examinations of the left side showed no abnormal findings.\nComputed tomography showed unilateral sphenoethmoidal sinusitis along with signs of orbital invasion. Most of the orbital lesions were located at the retrobulbar area and the orbital apex, and inferior and medial rectus muscles were thickened. Even though there was bony erosion of the posterior wall of the sphenoid sinus, there was no evidence of intracranial extension. Increased signal intensities were noted at the same regions on brain MRI with gadolinium enhancement (). The initial clinical diagnosis was OAS due to invasive fungal sinusitis. Therefore, we immediately started systemic amphotericin B (50 mg/day for 21 days and 25 mg/day for 10 days) and insulin therapy to control his blood sugar. The patient was also started on intravenous ampicillin/sulbactam (6.0 g/day) and ceftriaxone sodium (2.0 g/day) to cover any possible bacterial infection.\nThe patient elected to undergo endoscopic sinus surgery under local anesthesia on the 3rd hospital day. The sphenoid sinus was filled with thick mucus and brown-colored matted fungal balls. All the fungal materials were removed using curettes and sinus irrigation. The mucosa around the ostium and the fungal materials were sent to the Department of Pathology for the determination of invasiveness.\nBy the 4th postoperative day (POD), there was no improvement and the visual acuity decreased from finger count to light perception. The patient complained of severe headache, nausea and vomiting; the follow-up computed tomography and cerebrospinal fluid (CSF) study showed no evidence of meningitis or intracranial invasion. The histopathology showed aspergillosis without evidence of tissue invasion. Therefore, empirical corticosteroid therapy with prednisolone (PDS, 55 mg for 7 days, 40 mg for 7 days, and 20 mg for 7 days) was started on the 9th POD. After the 3rd day of PDS therapy, his headache and ophthalmic symptoms began to improve. With the exception of the ophthalmoplegia, the ptotic upper eyelid completely recovered and the visual acuity improved to 20/100 by the day of discharge (the 28th POD). The patient's blood sugar was not well controlled with fluctuations being noted during the hospitalization. By 3 months, the right visual acuity improved to 20/63 and the extraocular muscle movement was completely normalized. After 8 months, there was no recurrence of the nasal and orbital symptoms.