Familial occurrence of sporotrichosis is rare. We report two patients, a husband and wife, with sporotrichosis. A 67-year-old man and a 63-year-old woman, who were farmers, had erythematous plaques and nodules on their faces and his forearm. They had noticed their cutaneous lesions during nearly the same period without any traumatic history. Fragments of biopsy specimens from both of them were submitted for mycological cultures and yielded pure cultures of Sporothrix schenckii (S. schenckii). Restriction fragment length polymorphisms in the mitochondrial DNA of the S. schenckii isolated from the biopsy specimens of their lesions were investigated. The isolates were identified as type 5, which is comparatively abundant in the Kanto area in Japan. The husband was treated with potassium iodide and itraconazole. His wife was treated with itraconazole alone. We failed to isolate a causative fungus from the soil. Our case is the second case in the literature of sporotrichosis in a husband and wife during the same period.