Eur opean Rev iew for Med ical and Pharmacol ogical Sci ences 2010; 14: 491-493 A case of labial fusion and urinary pseudo-incontinence in an elderly woman. A surgical treatment and a review

L. PALLA, B. DE ANGELIS, L. LUCARINI, D. SPALLONE, G. PALLA*, V. CERVELLI

Plastic Surgery Department, University of Tor Vergata, Rome (Italy); *Operative Unit of Central Hospital of Viterbo (Italy)

Abstract. – Labial fusion is defined as ei - Urography, performed before the surgical ther partial or complete adherence of the time, revealed normal kidneys, ureteres, bladder minora (1), and also called vulvar fusion, adhe - and urethra morphology and reflux of urine in sions of the labia minor or conglutination of the the vagina (Figure 2). and sinechia of the . The com - plete and severe labial fusion is a rare pathology Preoperatively the woman was treated by with a small number of cases reported in the lit - oestriol 0.0125% cream daily for two weeks. erature in adults. Under a general anesthesia the labia were We present a case report of a postmenopausal separated by sharp dissection along the line of woman who presented with voiding difficulty labial adhesion to restore normal anatomy (Fig - and incontinence and was treated by surgical di - ure 3). vision of the adhesions and immediate resolu - tion of the urinary incontinence confirmed by A vesical 14 F catheter and vaginal pack were multichannel urodynamic test postoperatively. inserted in attempt to keep the raw area of the labia separated. The vaginal pack and vesical Key Words: catheter was removed at 2nd day postoperatively. Labial fusion, Urinary incontinence, Menopause. Oestrogen cream (oestriol 0.0125%) was topi - cally applied daily postoperatively to ward off a relapse. Removed the vesical catheter the woman was able to bladder voiding and to have urinary conti - nence. Multichannel urodynamic test postopera - Case Report tively was performed. The multichannel urody - namic test have shown: the bladder give a Con -

A 71-year-old woman with 3 vaginal deliver - tractions not inhibited (C.N.I.) 23 cm of H 2O; ies was referred with a 1-year history of symp - voiding normal pressures; moderate residue (25 toms of voiding difficulty and urinary inconti - ml); closing normal pressure; normal functional nence. Her past history included a vaginal hys - lenght; expelled volume: 314 ml; max flux: 18,5 terectomy and repair of cystourethrocele by ml/s; middle flux: 8,5 ml/s; voiding time: 39 s; means of Kelly-Kennedy technique in the 1995. flux time: 37 s; attainment time of max flux: 7 s; On examination of the vulva revealed that both normal sphinteric function. the labia minora were extensively fused. The labial adhesions covering the vaginal introitus, urethral meatus and . The rest of the vulva appeared atrophic (Figure 1); a pinhole opening Discussion at the midline. The woman was unable to have voiding stimu - Labial fusion is typically described in chil - lus. Urine was unable to escape freely through dren, especially in development countries, with the small introital opening and there was retro - highest incidence in the first 2 years of life 2,3 . grade filling of the vagina, which resulted in con - Labial fusion is a benign genital disorders in tinual leakage of urine postmicturition. girls: it may be either congenital or acquired,

Corresponding Author: Ludovico Palla, MD; e-mail: [email protected] 491 L. Palla, B. De Angelis, L. Lucarini, D. Spallone, G. Palla, V. Cervelli

Figure 3. Intraoperative views. Figure 1. Preoperatory view. presentation is 2.5 years, with more than 90% oc - sometimes due to poor hygiene. Congenital labi - curring under the age of 6 years 6. Parental panic al fusion may be associated with anatomical ab - about this “absent vagina” contrasts with its sim - normalities in the newborn including ambiguous ple, rapid, radical treatment 3. Is rarely reported in genitalia and congenital absence of the vagina or adolescents and women of reproductive age and occur as a result of true hermaphroditism, appears to be associated with a combination of pseudohermaphroditism, congenital adrenal hy - local inflammation and the estrogens deficiency perplasia or intrauterine exposure to exogenous seen in the premenarchal age group 4-7. One theo - androgens 4. Acquired labial fusion can occur also ry for labial fusion is low prepuberal estrogens for trauma to the upper squamous layer of the levels. In fact, spontaneous separation of labial labial epithelium with formation of scar tissue adhesions occurs at puberty with the production between the 2 opposed labia as healing occurs 5. of endogenous estrogens 1. Estrogen’s action in Acquired labial fusion can occur in childhood regard to collagen may influence recurrent adhe - in prepubescent girls and the commonest age of sions and adhesions that form after manual dis - ruption or surgical separation. Estrogens may have a role in vaginal healing in genital surgery. Topical estrogens remain the mainstay of ther - apy 8. Estrogens treatment – twice daily – is a long procedure (3 or 4 months) 3, 8. Lower urinary tract obstruction in women is an uncommon condition resulting from multiple either organic or functional disorders. Partial and incomplete or complete vulvar fusion usually oc - curs in significant repercussion on urination dy - namics 9. The etiology for the adhesions is unclear, al - though vulvovaginitis and mechanical irritation have been implicated as causative factors 6,12 . Chronic inflammation is thought to produce de - nudation of the thin surface epithelium, which in close approximation allows the labia to adhere to each other and result in obstruction of introitus 13 . The hypo estrogenic state may predispose the ep - ithelium to trauma and inflammation. The repro - ductive age, whit a normal sexual steroids produc - tion, may be protective against this condition 14,15 . Figure 2. The RX urography shown urine reflux in the A new surgical technique to treat refractory vagina. labial fusion in the elderly was presented in

492 A case of labial fusion and urinary pseudo-incontinence in an elderly woman

2) FISCHER GO. Vulval disease in pre-pubertal girls. Australas J Dermatol. 2001; 42: 225-234; quiz, 235-236.

3) GAUDENS DA, M OH -E LLO N, F IOGBE M, B ANDRE E, OSSOH BM, Y AOKREH JB, T EMBELY S, G OULY JC, ODÉHOURI T, O UATTARA O, DA -S ILVA -A NOMA S, K OBE - NAN RD. Labial fusion in the paediatric surgery department of Yopougon University hospital (Côte d’Ivoire): 108 cases. Sante 2008; 18: 35- 38.

4) ONG NC, D WYER PL . Labial fusion causing voiding difficulty and urinary incontinence. Aust N Z J Ob - stet Gynaecol 1999; 39: 391-393. Figure 4. Post-operative after 6 months. 5) CAPRARO VJ, G REENBERG H. Adhesions of the labia minora. A study of 50 patients. Obstet Gynecol 1972; 39: 65-69.

6) KLEIN VR, W ILLMAN SP, C ARR BR. Familial posterior 1989. This surgical technique is so described in labial fusion. Obstet Gynecol 1989; 73(3 Pt 2): detail: (1) Labial separation; (2) The subsequent 500-503. raw area is covered by a rotational skin flap from 7) CHUONG CJ, H ODGKINSON CP. Labial adhesions pre - the thigh. The flap is deroted of epithelium prox - senting as urinary incontinence in post - imally and tunneled subcutaneously. The distal menopausal women. Obstet Gynecol 1984; 64(3 portion emerged to cover the raw clitoral area Suppl): 81S-84S. and this successfully prevented contraction and 8) FINLAY HV. Adhesions of the labia minora in child - scarring. This new technique is suitable in elder - hood. Proc R Soc Med 1965; 58(11 Part 1): 929- ly patients with cases of labial adhesion that are 931. 16 refractory to all other treatment modalities . 9) QUEIPO ZARAGOZÁ JA, L ÓPEZ BAEZA F, B UDÍA ALBA A, We have reviewed the literature on labial fu - FUSTER ESCRIVÁ A, L LORET MARTÍ MT, J IMÉNEZ CRUZ JF. sion and when identified in the postmenopausal Infravesical obstructive uropathy secondary to ex - woman, these adhesions may be treated with a treme vulval atrophy. Actas Urol Esp 1999; 23: combination of surgery, topical hormones (estro - 792-796. gens and/or steroids) and manual separation of 10) HUFFMAN JW. Principles of adolescent gynecology. the labia during the reparative period with resolu - Obstet Gynecol Annu 1975; 4: 287-308. tion of coexisting urinary symptoms and dys - 11) CHRISTENSEN EH, O STER J. Adhesions of labia mino - function. Management of the incontinence and ra (synechia vulvae) in childhood. A review and agglutination and response to treatment were re - report of fourteen cases. Acta Paediatr Scand viewed. Medication usage was examined. 1971; 60: 709-715. In this case report the surgical treatment have re - 12) SCHOBER J, D ULABON L, M ARTIN -A LGUACIL N, K OW LM, solved the urinary symptoms and dysfunction as to PFAFF D. Significance of topical estrogens to labial prove by multichannel urodynamic test (Figure 4). fusion and vaginal introital integrity. J Pediatr Ado - lesc Gynecol 2006; 19: 337-339. The quick resolution of the urinary inconti - nence after the surgical lysis of the labial fusion 13) PULVINO JQ, F LYNN MK, B UCHSBAUM GM. Urinary in - should to define this condition a urinary pseudo- continence secondary to severe labial agglutina - tion. Int Urogynecol J Pelvic Floor Dysfunct 2008; incontinence and non-a urinary incontinence so 19: 253-256. as habitually is defined in the reviewed literature. 14) GOLDSTEIN AI, R AJCHER WJ. Conglutination of the labia minora in the presence of normal endoge - nous estrogen levels: an exception to the rule. Am References J Obstet Gynecol 1972; 113: 845-846.

15) KUO DM, C HUANG CK, H SIEH CC, L IOU JD, C HEN 1) BEN -A MI T, B OICHIS H, H ERTZ M. Fused labia. Clini - KC, H SIEH TT. Labial fusion in a thirty-year-old cal and radiological findings. Pediatr Radiol 1978; woman. Acta Obstet Gynecol Scand 1998; 77: 7: 33-35. 697-698.

493