A primary cutaneous lesion in a 5-year-old boy who had recently received chemotherapy for acute lymphocytic leukemia was found to be caused by a dematiaceous fungus, Drechslera spicifera. The lesion was an erythematous macule that rapidly developed necrotic ulcerations. The fungus, which is commonly found in soil and as a plant pathogen, was isolated from cultures of the lesion and from an excisional biopsy specimen. Hyphae and swollen hyphal cells resembling chlamydospores were observed in the biopsy specimen. Septate pigmented hyphae were found in the tissue, which is consistent with phaeohyphomycosis. Resolution of the infection occurred following excisional biopsy and systemic amphotericin B therapy. There was a concomitant recovery from neutropenia.