Leiomyoma of the female urethra is a rare condition. It is a benign mesenchymal tumor that commonly presents with recurrent urinary tract infections and various lower urinary tract symptoms. We report a case of urethral leiomyoma and review the literature. A 44-year-old woman presented with severe irritative voiding symptoms without urinary tract infection and bilateral upper urinary tract dilation. Ultrasound and cystoscopy revealed a smooth mass arising from the urethra and projecting into the bladder. Transurethral resection of the mass was performed and 32 g of tissue was removed. There were no complications and the symptoms resolved completely, indicating this to be a safe approach. Histopathology showed the tumor to be leiomyoma.