Two cases of protothecosis caused by Prototheca wickerhamii have been reported from Nagoya in a 12 year period in both cases the infection presented on the cheeks of otherwise healthy women. Biopsies showed numerous PAS positive staining organisms with the distinctive mulberry like endosporulation in the dermis. Prototheca wickerhamii was identified on sugar assimilation tests of colonies isolated from tissue on Sabouraud agar. Case 1 responded to 11 months of oral ketoconazole therapy. Case 2 might not respond to itraconazole. The source of the infections has not been identified.