We herein report a rare case of leiomyoma of the urethra. A young woman with no history of malignancy was referred to our hospital because of a 1-year history of frequent urination, urgency, and dysuria. Postoperative pathologic examination confirmed the diagnosis of urethral leiomyoma. After tumor resection, the patient was followed up for 2 months in the outpatient department and developed no obvious postoperative complications such as urinary incontinence.