The twenty-third case of penile leiomyosarcoma in the literature is reported, and all cases are reviewed. Clinically and pathologically these tumors fall into two groups, superficial and deep. Superficial tumors grow slowly, and tend to recur locally, but have a high salvage rate and good prognosis. The recommended treatment is local excision with negative margins. Recurrences are usually amenable either to re-excision or amputation with some degree of success. Deep tumors exhibit a more aggressive behavior, and have a poorer prognosis. Despite radical surgery the salvage rate is low, and most patients die from disseminated disease one or two years after diagnosis. Metastases to inguinal lymph nodes are uncommon, and should be treated aggressively in the absence of distant metastases. The role of adjuvant radiotherapy and chemotherapy in treating deep tumors should be investigated.