Abstract
Mucormycosis of the paranasal sinuses represents an important cause of morbidity and mortality in patients whose host defenses have been altered by primary disease or immunosuppressive therapy. The pattern of involvement by this fungus is changing with reports of mucormycosis occurring also in immunocompetent host. The involvement of isolated sphenoid sinus is rare. In the present case, the only presenting symptom was visual impairment. These changing trends in presentation, the extent, and the area of involvement are challenging for the otorhinolaryngologist, ophthalmologist, and neurosurgeon. High index of suspicion, prompt intervention, and aggressive therapy are required to reduce the morbidity and mortality associated with this disease.
1
Introduction
Mucormycosis is an aggressive fungal infection caused by ubiquitous mould genera such as Mucor and Rhizopus that cause furry coatings on food. In healthy people, the immune system prevents infection from inhaled spores; however, in immunocompromised individuals, the mould can grow rapidly . Diabetes, trauma, immunodeficiency, immunosuppressive therapy after transplantation, renal failure, and also the use of desferoxamine are considered to be the major predisposing factors . Sinonasal mucormycosis is not common and rarely infects a healthy host. The presenting symptom and physical finding are often subtle . The exact pathogenesis of rhinocerebral mucormycosis and its pathways of spread are not clearly known. It is generally believed that mucor initially inoculates the nasal mucosa, spreading to the paranasal sinuses (specifically the ethmoid and maxillary sinuses), orbit, and finally the intracranial fossa . Isolated sphenoid sinus involvement is rare in mucormycosis. We came across 3 cases of isolated sphenoid sinus involvement in the literature, but unlike present case, 2 cases reported by Chopra et al had other symptoms along with visual impairment, whereas in our case, visual impairment was the only presenting symptom. In the third case by Chol et al , presenting symptoms have not been specified.
We report here a case of isolated sphenoid sinus mucormycosis having visual impairment as the only presenting symptom. The patient was successfully managed by endoscopic sphenoidotomy and debridement followed by amphotericin B and itraconazole administration. Posttherapy, the patient had a radiologic evidence of disease-free sphenoid sinus.
2
Case report
A 71-year-old patient presented with blurring of vision left side of 1 month duration. The patient was referred from the ophthalmology department, when the ophthalmologist could not ascertain any ophthalmologic cause for blurring of vision. There was no history of any medical illness or immunosuppressive therapy. Patient was found to be diabetic on routine hematologic examination and was started on insulin. Routine otorhinolaryngologic examination was unremarkable. Finger counting both eyes was more than 3 feet, but there was blurring of vision on the left side. Both pupils were normal in size and reaction. Computed tomographic (CT) scan of nose and paranasal sinuses revealed a soft tissue density localized only to the left sphenoid sinus, the and rest of the paranasal sinuses were clear ( Fig. 1 ). Based on the history, clinical examination, and radiologic findings, either a fungal etiology or malignancy was kept as a possible differential diagnosis. Because of the deterioration of vision, an urgent sphenoidotomy and biopsy were planned. Intraoperatively, the sphenoid sinus was full of cheesy and gritty material. Postoperative histopathologic examination was consistent with mucormycosis. Patient was started on injection amphotericin B 75 mg per day with regular monitoring of blood glucose levels and serum electrolytes especially serum K + . The blurring in vision started improving 2 weeks postoperatively. After completion of 2500 mg of amphotericin B, a repeat CT scan revealed complete resolution of disease from sphenoid sinus with completely normal vision ( Fig. 2 ).
Because radiologic evidence of clearance of disease was present, amphotericin was stopped and oral itraconazole was started in the dose of 200 mg twice a day for 3 months. The patient was monitored throughout the therapy for hepatotoxicity. The patient is on regular follow-up and is disease-free for the last 6 months.
2
Case report
A 71-year-old patient presented with blurring of vision left side of 1 month duration. The patient was referred from the ophthalmology department, when the ophthalmologist could not ascertain any ophthalmologic cause for blurring of vision. There was no history of any medical illness or immunosuppressive therapy. Patient was found to be diabetic on routine hematologic examination and was started on insulin. Routine otorhinolaryngologic examination was unremarkable. Finger counting both eyes was more than 3 feet, but there was blurring of vision on the left side. Both pupils were normal in size and reaction. Computed tomographic (CT) scan of nose and paranasal sinuses revealed a soft tissue density localized only to the left sphenoid sinus, the and rest of the paranasal sinuses were clear ( Fig. 1 ). Based on the history, clinical examination, and radiologic findings, either a fungal etiology or malignancy was kept as a possible differential diagnosis. Because of the deterioration of vision, an urgent sphenoidotomy and biopsy were planned. Intraoperatively, the sphenoid sinus was full of cheesy and gritty material. Postoperative histopathologic examination was consistent with mucormycosis. Patient was started on injection amphotericin B 75 mg per day with regular monitoring of blood glucose levels and serum electrolytes especially serum K + . The blurring in vision started improving 2 weeks postoperatively. After completion of 2500 mg of amphotericin B, a repeat CT scan revealed complete resolution of disease from sphenoid sinus with completely normal vision ( Fig. 2 ).
