Case Presentation:

A 27–year–old male with unremarkable past medical history presented to the hospital with severe headache for two days. He had associated sinus pressure, nasal congestion with purulent discharge, proptosis, and tearful eyes for three months. Blood pressure was 153/89 mmHg, temperature 97.8°F, heart rate 70 beats/min, respiratory rate 20/min, and pulse oximetry 100% on room air. Physical examination showed a large polyp obstructing the left nostril. WBC was 7300/mm with 39% neutrophils, 49% lymphocytes, 10% eosinophils. He had negative blood cultures and HIV serology. Magnetic resonance imaging revealed large polypoid mass in left nasal cavity extending into left frontal, maxillary and ethmoid sinuses. There was a mass effect on left orbit causing deviation of left medial orbital wall with proptosis. Subdural 14mm empyema compressed the left frontal lobe and lateral ventricle. He was empirically started on antibiotics for bacterial infection and underwent surgical removal of polyp with endoscopic sinusotomy and left orbital decompression. Specimens showed fungal elements. Antibiotics were discontinued and patient was started on voriconazole. Periodic acid Schiff stain demonstrated fungal hyphae characterized by nonparallel walls and scattered septation. Cultures of three separate areas grew dark fungus identified as Bipolaris spicifera. Subsequent MRI showed improving aeration of paranasal sinuses and extra axial fluid collection in anterior cranial fossa consistent with epidural abscess. Left frontal craniotomy with drainage and removal of granulation tissue was performed. Patient responded well to surgery and antifungal medication. He was discharged on voriconazole with outpatient follow–up.

Discussion:

Bipolaris spicifera is part of the large Bipolaris group of dematiaceous fungi and is usually found in plants and soil. Previously relatively unknown, it is now being recognized with increasing frequency as a cause of human disease. Bipolaris spicifera usually causes sinusitis but may also cause meningitis, fungemia, peritonitis and endocarditis. Increasing numbers of cases of disseminated disease parallels the increase in immunocompromised patients, especially AIDS and transplant recipients. Fungal inhalation can result in the growth of sinus masses. Increased response of eosinophilic granulocytes against fungi rather than allergy (IgE mediated) is the proposed mechanism of this chronic inflammation. The polyps that develop require surgical removal followed by systemic antifungal therapy with itraconazole or voriconazole and systemic and topical steroid therapy.

Conclusions:

Bipolaris spicifera causes invasive form of sinusitis. Immunologic mechanisms are considered to be the underlying cause. Treatment plan incorporating medical, surgical and immunologic care remains the most likely means of providing long–term control of disease.