Use of atresic vagina as a urethra to repair iatrogenic incontinence, secondary to a complete longitudinal urethral incision

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Blackwell Science, LtdOxford, UKIJUInternational Journal of Urology0919-81722005 Blackwell Publishing Asia Pty LtdMarch 2005123325327Case ReportUse of atresic vagina as a urethraS Albayrak et al.

International Journal of Urology (2005) 12, 325–327

Case Report

Use of atresic vagina as a urethra to repair iatrogenic incontinence, secondary to a complete longitudinal urethral incision SELAMI ALBAYRAK, CEMAL GÖKTAS AND ÖNDER CANGÜVEN Kartal Teaching Hospital, Second Urology Clinic, Kartal-Istanbul, Turkey Abstract

Vaginal atresia is a rare congenital anomaly. Patients with this pathology commonly present with amenorrhea, or an inability to have sexual intercourse. A dorsal urethral wall incision was performed in a woman who complained of difficulty with coitus. Her gynecologist intended to expand the vagina, thinking the diagnosis was a vaginal septum. We report a repair technique for total urinary incontinence following a longitudinal complete urethral incision, which also involved the bladder neck. The vaginal reconstruction consisted of an ileal segment, whereas the urinary incontinence was corrected using a rectus fascial sling. To our knowledge, this is the first case of this kind reported in the literature.

Key words

amenorrhea, atresia, ileum, urethra, vaginal injury.

Introduction Vaginal atresia is a rare congenital anomaly. Patients generally present with amenorrhea and/or an inability to have coitus. The patient in the current study presented to a gynecologist with the latter compliant. Unfortunately, the gynecologist incised her urethra at the six o’clock position because of misdiagnosis of vaginal septum. As a result, the patient was referred to us for total urinary incontinence. We report the present case with a management strategy and outcome.

Case report A 25-year-old woman presented with a 6-month history of urinary incontinence. She was married 6 months previously and had had difficulty with sexual intercourse. The patient underwent a surgical procedure for repair of a presumed vaginal septum, but became totally incontinent following the procedure. The patient had primary Correspondence: Önder Cangüven MD, Sakaci sokak Yildiz, Apt. 42/5, Erenköy, TR-81090 Istanbul, Turkey. Email: [email protected] Received 8 March 2004; accepted 13 September 2004.

amenorrhea. On physical examination, the patient was phenotypically female and had well-developed secondary sexual characteristics. During the genital examination, her vagina was noted to be very narrow. It would not permit a speculum. The introitus was incompletely atretic, with a 3–4 cm distal ectodermic segment. The external urethral meatus could not be found. On endoscopic examination, the upper wall of the vagina had an incision extending posteriorly (Fig. 1). Urine passed directly to the vagina from the bladder neck during assessment. The rest of the bladder exam was normal. All laboratory results were normal. A computer tomography (CT) scan showed that the patient’s ovaries were atretic and a uterus could not be detected. There were no additional urinary system anomalies. Management

We performed a subumbilical midline incision. Once the patient’s bladder was opened, we noted that the distal portion of the trigone was in continuity with her vagina. The atretic vagina and incised urethra were kept in continuity and were used as a neourethra. A fascial segment 2-cm wide and 15-cm long was removed from autologous rectus muscle. Using blunt dissection with an index finger, the space between the vagina and rectum

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Fig. 1 Preoperative appearance. White arrow, incised floor of urethra; black arrow, atresic vagina; transparent arrow, clitoris.

Fig. 3 Postoperative appearance. White arrow, neovagina; black arrow, neourethra.

Discussion

Fig. 2 Autologous rectus fascia sling was passed twice around the bladder neck and posterior vagina wall, and then fixed to Cooper’s ligament.

was developed. The rectus fascia sling was passed twice around the bladder neck and posterior vaginal wall to form a double loop. It was fixed to Cooper’s ligament using Prolene sutures (No. 0) on each side (Fig. 2). After closure, the bladder was filled with 300 cc of isotonic saline solution. There was no leakage at the level of the bladder neck. However, it was possible to pass urine with Crede’s maneuver. Following this, a 15-cm long ileal segment was removed 40-cm proximal to the caecum. The proximal end of this segment was closed with 3/0 vicryl (Polyglactin 910) and the distal end was mobilized and anastomosed to the introitus with 2/0 vicryl through the endopelvic fascia (Fig. 3).

Vaginal atresia is a congenital anomaly that is seen in 1/4000–10 000 females.1 This müllerian duct developmental anomaly (Mayer–Rokitansky–Küster–Hauser Syndrome) results in congenital absence of a uterus and vagina. In this syndrome, there is a lack of müllerian development, a congenital absence of the vagina and a rudimentary uterus, with normal uterine tubes, ovaries, and secondary female sex characteristics and normal growth. Patients generally present with amenorrhea and/ or an inability to achieve coitus. Although our patient was amenorrheic, her main complaint was an inability to achieve coitus. The patient’s gynecologist diagnosed the patient as having a vaginal septum, believing that her urethra was a part of that septum. Consequently, the urethra was incised along the urethral wall up to the bladder neck at the six o’clock position. The patient subsequently developed total urinary incontinence. The internal sphincter is the most important part of the mechanism in female micturition physiology. Complete urethral division is a rare complication. Injuries of female urethra are generally traumatic,2,3 and are seen after urologic and gynecologic pelvic surgeries.4 To our

Use of atresic vagina as a urethra

knowledge, there are no published accounts of cases similar to that reviewed in the present case. Therapeutic choices for this patient were particularly confusing, because the incision extended to the bladder neck. There is not a standard technique for repairing such an injury. Possible surgical procedures include: (i) Placement of a permanent cystostomy or intermittent catheterization with a Mitrofanoff procedure after closure of bladder neck;5 (ii) creating a conduit like stoma between the bladder and skin; and (iii) clean intermittent catheterization following a type of sling repair to secure continence by using the atretic vagina as a urethra. Considering the patient’s age and social needs, we preferred the last choice. On Day 3 after the operation, the patient’s transurethral catheter was removed and the patient performed clean intermittent catheterization for 1 week. Sixth months after the operation, she continued to achieve controlled spontaneous micturition, kept dry and had no residual urine. She was also sexually active. We recommend that this technique be applied when such a complication is encountered.

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References 1

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American College of Obstetricians and Gynecologists Committee Opinion. Non surgical diagnosis and management of vaginal agenesis. Obstet. Gynecol. 2002; 100: 213–16. Hemal AK, Dorairajan LN, Gupta NP. Posttraumatic complete and partial loss of urethra with pelvic fracture in girls. An appraisal of management. J. Urol. 2000; 163: 282–7. Desai RM, Sabnis RB, Bapat SD. A new outlook in the management of complex female urethral injuries. Arch. Esp. Urol. 1993; 46: 749–51. Mattingly RF, Borkowf HI. Acute operative injury to the lower urinary tract. Clin. Obstet. Gynaecol. 1978; 5: 123–49. Leonard MP, Gearhart JP, Jeffs RD. Continent urinary reservoirs in pediatric urological practice. J. Urol. 1990; 144: 330–3.

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