Objective. Report of first case of Protothecosis zopfii in China and causes the skin infection in the world. Method.By clinical and laboratory examinations to confirm the diagnosis and the response to treatment. By the review of literatures to confirm the first case of human skin infection in the world. Result.From the literatures and the clinical pictures

it is confirmed that this is the first case report of Protothecosis zopfii of skin in the world. Conclusion.The first case of Protothecosis zopfii in human being was reported and successfully treated with local infiltration of Diflucan （fluconazole） 3ml （2mg/ml）/week×4.