Basidiobolomycosis is a deep mycosis which preferentially affects rural young people in tropical countries. We report a case of basidiobolomycosis successfully treated with ketoconazole. It was a 9-year-old boy of rural origin in whom the diagnosis of basidiobolomycosis was suspected due to a deep skin infiltration involving the chest and neck. Histology revealed hypodermic granulomatous inflammation with predominantly macrophage and eosinophils. The child was treated successfully with ketoconazole in eight weeks. Treatment of basidiobolomycosis is based on azole derivatives which are particularly effective. Histopathology is very important in the diagnosis of this affection, especially in tropical countries where it may simulate Mycobacterium ulcerans infection.