We report a 71-year-old Japanese healthy female with an unusual clinical course of sporotrichosis; she developed infectious lesions on the face and the left upper limb at different periods. The initial lesion appeared on her right cheek and nose in 1992. Histopathologic and fungal examinations comfirmed the diagnosis of sporotrichosis. She was treated with oral potassium iodide and/or itraconazole and topical heat therapy. She had three recurrences and her facial plaque, which was resistant to several conventional treatments, was eventually excised and then grafted in 1996. After the surgical procedure, oral medication was continued for two months. In 2001, new lesions appeared on her left hand and forearm, which were anatomically different from the former lesions. Based on histopathologic and mycological findings, we confirmed reinfection with sporotrichosis. She was again treated with potassium iodide. The cutaneous lesions completely resolved after 26 weeks without surgical treatment.