Leiomyoma is a benign tumor that originates from smooth muscle. It is extremely rare in the urethra, with approximately 150 reported cases. The uterus is the most common site in women of reproductive age (1), and it is more common in women in the third and fourth decade of life (1-3). The symptoms depend on the location and size of the lesion (4). It usually affects the proximal urethra, and the mean diameter is 3.7 cm (2,5,6). The main signs and symptoms are mass sensation in the urethra, dyspareunia, storage and voiding symptoms of the urinary tract, hematuria, and acute urinary retention (1,3,5). Imaging tests such as ultrasound and magnetic resonance imaging (MRI) are important to help diagnose and adequate surgical planning (1,3). The anatomopathological evaluation is essential to differentiate it from leiomyosarcoma (1,3). There is no report of malignant transformation (1,2). Complete surgical excision of the lesion is the treatment of choice (1-6). Lesion recurrence is rare, with only three reports in the literature, and the treatment chosen was surgical re-approach with complete lesion resection (1,7,8).

We present the surgical excision of urethral leiomyoma in a stepwise video of a case report.

A 39-year-old female patient with a complaint of a periurethral nodule. She had a previous diagnostic hypothesis of a skene cyst, and the lesion was resected four years ago, and the anatomopathological revealed leiomyoma. She noticed the recurrence of the lesion with progressive growth for the last year, and with some voiding effort, weak urinary stream, and dyspareunia. Then she was referred to our urology team. The physical evaluation showed a large nodule in the supra urethral region, with a displacement of the urethral meatus downwards. A pelvic MRI showed an expansive nodular lesion, measuring 2.6 x 2.2 x 2.4 cm, with well-defined limits, located in the distal third of the urethra. It involved the anterior urethral wall, between 3 and 9 hours. We proposed a surgical resection. The procedure took place in a lithotomy position under a spinal block. We made a supra meatal incision and dissected the lesion dorsally and laterally. The posterior aspect of the lesion had close contact with the urethra, and we dissected carefully to preserve the urethra. However, it was not possible at some point due to a suspected invasion of this area. Thus, we reconstructed the urethra in two planes of a continuous suture with 5-0 polydioxanone threads. Then, we reconstructed the pubourethral ligaments with one stitch of 4-0 polyglactin threads in each side of the urethral, to reestablish the urethral support and anatomical positioning. She was discharged on the first postoperative using a foley catheter, which was removed on the fourteenth day. She could void with no difficulty and had no urinary incontinence. At a six-month follow-up, she had a topical meatus with no signs of retraction or displacement. The anatomopathological evaluation revealed atypical leiomyoma (symplastic), with degenerative atypia, absence of necrosis, absence of mitotic figures, low proliferative index (Ki-67: 6.99%), and a free surgical resection margin. Patients with compromised margins have a higher risk of recurrence, just like our patient had during the first surgery. Lesion recurrence requires new excision, and there is no indication for adjuvant radiotherapy.

Urethral leiomyoma is a rare pathology and can present with an abnormal voiding, and there is no established protocol for its treatment. The challenge is to resect the lesion entirely while preserving the structures. It is crucial to reconstruct the urethra and connective tissue support to maintain adequate urinary function.

Jiménez Navarro M, Ballesta Martínez B, Rodríguez Talavera J, Amador Robayna A. Recurrence of urethral leiomyoma: A case report. Urol Case Rep. 2019 Jul 16;26:100968. doi: 10.1016/j.eucr.2019.100968. PMID: 31367525; PMCID: PMC6656683.
Popov SV, Orlov IN, Chernysheva DY, Grin' EA. Urethral leiomyoma: A rare neoplasm. Urol Ann. 2021 Apr-Jun;13(2):194-197. doi: 10.4103/UA.UA_90_20. Epub 2021 Mar 4. PMID: 34194152; PMCID: PMC8210724.
Fedelini P, Chiancone F, Fedelini M, Fabiano M, Persico F, Di Lorenzo D, Meccariello C. A very large leiomyoma of the urethra: A case report. Urologia. 2018 May;85(2):79-82. doi: 10.5301/uro.5000223. Epub 2017 Feb 28. PMID: 28256703.