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We describe a case of extensive human cutaneous protothecosis in the northeastern of Brazil. The diagnosis was made through histopathological examination of skin samples and culturing on Sabouraud dextrose agar, with identification through mass spectrometry. Treatment with itraconazole failed and was switched to liposomal amphotericin followed by voriconazole with clinical cure. Prothotecosis should be a diagnostic hypothesis when there are skin lesions after trauma, specially in situations of atypical evolution.