A 12-year-old girl had been presenting a woody infiltration and erythema in the frontal region and on the entire left half of the face, leading to deformity of the nose and buccal fissure, and adenomegaly in a posterior cervical chain, for the last 18 months. Sinusitis was diagnosed and treated with antibiotics, and submitted to ethmoid sinusotomy, with no improvement. Several laboratory tests were made to find the correct diagnosis. An intradermal test for delayed hypersensitivity showed a positive reaction (5 mm) with necrosis for metabolic antigens for Conidiobolus. An oral mucosa biopsy showed a dense lymphohistiocytic infiltrate and focal points of necrosis. Gomori staining for fungi revealed countless wide, nonseptate hyphae. Amphotericin B was prescribed during 35 days, with no improvement. Terbinafine given orally was started in association with amphotericin B. Reduction of the lesions was observed 2 months later. No side effects were seen during 4 months of treatment.