Background: A leiomyoma is a benign tumor of smooth-muscle cells that can arise from the genitourinary or gastrointestinal systems. Leiomyomas arising from the urethra or paraurethral areas are uncommon, and there are few reported cases. Case: A 25-year-old female presented with a painless paraurethral swelling during her first trimester of pregnancy. This swelling progressively increased in size, causing obstructive urinary symptoms. Examination under anesthesia was performed, and diagnostic cystoscopy-urethroscopy was performed prior to surgical excision of the mass to ensure that the site of origin had no urethral communication. Results: Histopathologic evaluation confirmed the diagnosis of a leiomyoma. No postoperative complications were noted, and the patient's urinary symptoms were relieved. Conclusions: It is important to differentiate a urethral leiomyoma that presents mainly with obstructive urinary symptoms and requires surgical endoscopic resection from a paraurethral leiomyoma that presents mainly as an asymptomatic freely mobile lump and does not communicate with the urethra, having a clear line of cleavage in between. ( J GYNECOL SURG 36:144