A case of urethral leiomyoma is presented. The patient was a 48-year-old female who was hospitalized with a 4-year history of a progressively enlarging urethral mass. Based on clinical diagnosis of benign urethral tumor, local excision was performed. Histological diagnosis was urethral leiomyoma and the tumor cells demonstrated immunoreactivity for estrogen receptors. We report our experience of a leiomyoma of the urethra expressing estrogen receptors.