Pseudoallescheria boydii, a fungus often isolated from soil, commonly causes a mycetoma. Successful treatment involves resection of the infected area and appropriate antifungal chemotherapy. In vitro sensitivity of P. boydii to miconazole is usually demonstrated, but resistance to amphotericin B and 5-fluorocytosine is often observed. Limited data are available on the treatment of this infection with ketoconazole. An usual case of nontraumatic P. boydii soft tissue infection without draining sinus tracts occurred in a 50-year-old woman. Nontraumatic P. boydii soft tissue infection was diagnosed by cytologic preparation and fungal cultures of the aspirate. The patient was successfully treated with en bloc resection for four months of oral ketoconazole.