To report one case of urethral angioleiomyoma because of the rare site, and to review its main characteristics.We report one case of male urethra angioleiomyoma presenting with obstructive symptoms.We performed excision and end to end anastomosis of a small bulbar urethra stenosis. Pathology reported angioleiomyoma.Angioleiomyoma is a benign tumor arising from smooth muscle within blood vessel walls. It is extremely rare in the urethra. It is more common in females. Recurrence and metastasis are exceptional. It shows scarce or moderate cellularity with predominance of fusiform cells. They may express smooth muscle vimentin, desmin, and actin. The best therapeutic option is resection.