Leiomyoma is a benign smooth muscle tumor which is rarely found in urethra. It often appears in females during their reproductive age (from menarche to menopause); the mean age of their appearance is approximately 41 years. Less than 100 cases were reported in the literature.We hereby report a case of a 52-year-old White woman who presented with complaints of dysuria and urinary tract infection. The mass was completely removed by transvaginal excision with a rim of normal tissue. Histopathological studies confirmed the urethral leiomyoma. The patient remained asymptomatic and there was no evidence of recurrence in the followup.