Leiomyomas are benign tumors of smooth muscle origin and are very rare in the female genital tract. To date, approximately 35 cases of urethral leiomyoma have been reported in the literature. A 34-year-old woman presented with a 3-year history of a mass at the urethral meatus. Physical examination showed 2 x 1.5 cm lump at the urethral meatus, posterior lip. Histologically the tumor was mainly composed of benign cigar shaped smooth muscle izells which were arranged in interlacing fascicles without cellular atypia or mitosis. Immunohistochemistry confirmed leiomyoma with positive staining for vimentin, desmin and muscle specific actin.