A case of leiomyosarcoma of the penis is reported. A 27-year-old-man presented to our department with a mass at the root of the penis. Biopsy of the tumor showed that the tumor was leiomyosarcoma. The tumor was clinically and pathologically categorized into the deep type. Despite total penectomy and adjuvant chemotherapy, the patient died from disseminated disease 14 months after surgery. This is the 45th case of penile leiomyosarcoma.