Introduction: The leiomyoma of the urethra is rare. It is a benign mesenchymal tumor that affects the smooth muscle of the urethra. These tumors often occur in women of childbearing age the middle age of their appearance is about 41 years old. Case report: Here we report the case of a 37 years old white woman with progressive dysuria with nodule sensation in the vagina Conclusions: Leiomyoma is a benign tumor, common in the uterus but very rare in the urethra. His diagnosis is rarely mentioned. The para-clinical assessment specifies the characteristics, the reports of the tumor, and directs on its nature. Open or endoscopic surgical excision allows both the diagnosis and the treatment of symptoms.