The authors reported the case of a symptomatic HIV-infected woman with a slowly progressive infiltrative lesion which invaded in and around the nasal cavity over a 6-month period. Physical examination showed erythematous to violaceous plaques at the nasal and malar areas. Swelling of the inferior turbinate was noted in the right nare. Skin biopsy of the involved area revealed multiple nonseptate, broad, thin-walled hyphae within giant cells and granulomata. Entomophthoramycosis was diagnosed based on clinical features and histopathology. She was treated with intravenous amphotericin B for two weeks, followed by oral itraconazole 400 mg daily. At six months there was complete resolution of all lesions.