New attempt using labio-vestibular flap technique to manage circumcised women with Rokitansky syndrome

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1 Acta Obstetricia et Gynecologica. 2008; 87: 9498 ORIGINAL ARTICLE New attempt using labio-vestibular flap technique to manage circumcised women with Rokitansky syndrome SAEED MOHAMED AHMED THABET 1 & ATHMAR HUSSEIN ALI 2 1 Cairo University, Egypt, and 2 Sana a University, Yemen Abstract Objective. To assess the efficacy of the labio-vestibular flap technique in managing circumcised women with Rokitansky syndrome and in correcting the sexuality defects caused by the anomaly present and by female circumcision. Design. A prospective, comparative, clinical study. Setting. Kasr El Aini School of Medicine, Cairo University, Egypt. Patients. Thirty-four circumcised patients with Rokitansky syndrome were divided into 2 groups; Group 1 consisted of 22 cases treated with the labio-vestibular flap technique, and Group 2 consisted of 12 cases treated with McIndoe s technique. Main outcome measures. Pre and postoperative clinical assessment of the newly formed vagina and sexuality. The new flap is formed of the remaining parts of the labia minora and the adjoining parts of the vestibule, and is used to cover the posterior and lateral walls. Meanwhile, the anterior wall is covered by the epithelium of the original blind vaginal pouch. The modified Kasr El Aini sexual assessment sheet assesses sexuality. Results. Rokitansky syndrome could be classified into 4 clinical types according to the degree of development of the uterus. The labio-vestibular technique was simple and not associated with any graft rejection, hair growth or contraction of the reformed tract. Dyspareunia and marital failure were less significantly recorded in the labio-vestibular technique than in McIndoe s technique. But, the most significant results were the improvement in sexuality, in addition to restoration of genital continuation, menstruation and fertility in some cases. Conclusion. The labio-vestibular flab technique is the most simple and most suitable line of treatment for circumcised cases with Rokitansky syndrome. In these cases, the technique achieved good results in correcting sexual defects after circumcision. Key words: Rokitansky syndrome, female circumcision, neovagina, labio-vestibular flap Introduction Reconstruction of the vagina is an essential step in the restoration of sexuality in patients with Rokitansky syndrome. Sexuality is known to be maximally involved in these cases (1). The vagina is definitely absent in all cases, but the ovaries are commonly well developed and functioning. Meanwhile, development of the tubes and uterus is variable. In addition, female circumcision is associated with variable defects in the clitoris and labia minora. Accordingly, these defects of the vagina and vulva are expected to be associated with more defects in sexuality than other non-circumcised cases. Many techniques are used in the reconstruction of a new vagina; probably the most common technique is that by McIndoe and Banister (2). All techniques include creating a space between the urethra bladder complex and rectum, and epithelisation of that space by split-thickness skin, intestinal mucosa, peritoneum, amniotic membrane, and maybe other material (3). Other less popular techniques were also described (4,5). The main disadvantages of the former group are the rejection of the epithelising graft, contraction and stenosis of the created canal and lake of sexual sensitivity due to the lake of sexual nerve endings in the used graft (6). In addition, most of these operations do not include a step for establishing utero-vaginal connection in cases with a functioning or non-functioning but intact uterus. Recently, two other techniques have been used because of their simplicity and their effect on correcting sexuality; these include the use of the Correspondence: Saeed M.A. Thabet, 19 Maamal Al-Alban St. El-Kalafawi, Shoubra, Cairo, Egypt. dr_saeedth@hotmail.com (Received 23 October 2007; accepted 28 October 2007) ISSN print/issn online # 2008 Taylor & Francis DOI: /

2 The circumcised Rokitansky cases 95 mucosa of the vestibule of the vagina (7), and the labia minora skin flap (8) for the supposed vaginoplasty. Unfortunately, for most circumcised women, these techniques may not be suitable. Female circumcision is a problematic custom practiced in many countries in Africa and Asia. It includes partial or complete removal of the clitoris and labia minora, accordingly the labia minora graft needed for epithelisation of the newly created vagina in Rokitansky syndrome may be a problem. The authors suggest a new flap technique made of the remains of the labia minora, in addition to parts of the mucosa of the vestibule, to suit these cases of circumcised women. This study was carried out to assess the labiovestibular flap technique as a simple method for epithelising a newly formed vagina and to determine the role of this technique in correcting sexuality in circumcised cases of Rokitansky syndrome. Patients and methods Thirty-four cases of Rokitansky syndrome managed at Kasr El-Aini School of Medicine in the last 7 years were included in this study. Diagnosis was proved by the standard clinical findings and known investigations. All cases were aged 1924 years and married for 318 months. The preliminary sexual assessment was performed at the initial visit by the modified Kasr El-Aini sexual assessment questionnaire sheet (9), and the post-operative reassessment was obtained at the end of the follow-up clinical assessment. The latter was carried out every 3 months for 24 months postoperative. At each visit, the depth, diameter and state of the neovaginal lining were determined. The modified sexual assessment sheet includes 75% of the scores of the original sheet, in addition to the husband scores, which added 25 points to make a total score of 100. The husband scores are based on the ability of the husband to identify the anomaly present, the acceptability and the male desire after identifying the anomaly, the response of the patient to sexual stimulation from the point of view of the husband, the husband s sexual satisfaction after the operation, and the presence of any sexual complaint in the husband. The cases studied were divided into 2 groups and were managed by the classic technique of McIndoe (12 cases), and the newly modified technique of the labio-vestibular flap suggested by the authors (22 cases). The latter technique was carried out in three steps and performed in one operation. The steps were: Step 1: creating an avascular space between the urethra and the bladder complex and rectum for the neovagina. Step 2: epithelising the anterior wall of the neovagina using the epithelial lining of the blind vaginal pouch after its separation from its posterior attachment using a U-shaped incision. A transverse cut of the U-shaped incision was performed to separate the vaginal lining from the perineal skin (Figure 1); meanwhile, vertical cuts of the same incision were performed to separate the anterior lining from the posterior at the side walls of the pouch. The posterior flap was completely separated from its posterior attachment up to the dome of the pouch where dissection was completed to the level of the supposed cervix or its ligaments or a depth of 8 10 cm (Figure 2). Then, the separated flap was fixed essentially to the sides of the perivesical fascia as high as possible, and to the cervix or its ligaments when they could be identified. Step 3: epithelising the posterior wall of the neovagina using two labio-vestibular flaps formed by the remaining parts and bases of the labia minora and the adjoining parts of the vestibular epithelial lining (Figure 3). These flaps were created using inverted U-shaped incisions around the necessary sites and according to the length and width needed to cover the posterior wall and most of the sides of the newlyformed vaginal tract. These flaps were then fixed to the sides of the prerectal fascia as high as possible, and to the cervix or its ligaments when they could be identified (Figure 4). The latter made vagino-cervicouterine continuity easily achievable in most cases. A vaginal mould was not needed in any of these cases, but a vaginal pack was fixed for 2448 h. When haematomata was detected ultrasonographically, Figure 1. Separation of the mucosa of the blind vaginal pouch from its posterior attachment.

3 96 S.M.A. Thabet and A.H. Ali Figure 2. Elevation of the separated mucosal flap and dissecting of the space for the new vagina. continuity could be postponed until the graft was taken. All parts of the research design were revised and approved by the Institutional Review Board (IRB) and by the ethics committee at Cairo University. In addition, all cases consented to participation in this study, and the required surgery was explained before the operation. Results Rokitansky syndrome was identified and classified into 4 grades. Grade I (4 cases; 11.8%), the segmental absence of the vagina in the presence of a functioning uterus. Grade II (7 cases; 20.6%), the absence of the vagina in the presence of a hypoplastic uterus. Grade III (10 cases; 29.4%), the absence of the vagina in the presence of rudimentary uterine horns. Grade IV (13 cases; 38.2%), the absence of the vagina with total absence of the uterus. All cases were circumcised women Grade II, with a Figure 4. Complete epithelisation of the neovagina. clitoridal length of B1 cm and a labia minora width of B1 cm. No intraoperative or other early postoperative complications were recorded. All patients were sexually active 1 month after the flap technique and 3 months after McIndoe s technique. In the cases managed by the flap technique the mucosa of the vagina was pink, trophic and moist at the end of the first postoperative month and some vaginal folds Figure 3. Fashioning the labio-vestibular flap after full epithelisation of the anterior wall. Figure 5. The appearance of the vulva at the end of the labiovestibular flap technique. Notice the indistinguishable vulvar appearance and the vaginal pack used for 2448 h after the operation.

4 Table I. Results of the operative management of Rokitansky syndrome in circumcised women. The circumcised Rokitansky cases 97 Technique Labio-vestibular flap (22 cases) No. % McIndoe (12 cases) No. % Rejection of the graft Contraction of the neo-vaginal tract Dyspareunia Hair growth in the reconstructed tract Restoration of the genital continuity Restoration of menses Restoration of sexuality Sex scores before operation Sex scores after operation $ % Restoration of fertility No. of divorced cases $Significant postoperative increase of scores (T/df58.48/42 and pb0.005). %Significant postoperative increase of scores (T/df21.54/22 and pb0.05), but this increase was significantly less than that of the flap technique (T/df47.32/32 and pb0.005). were also noticed. Two fingers were introduced easily into the neovagina in all cases, and the mean length was cm. On the other hand, cases managed by McIndoe s technique showed the vaginal mucosa to be less vascular, thin and less moist with no vaginal folds. The vagina could admit two fingers and the mean length was cm. Rejection of the grafts, contraction of the neovaginal tract and hair growth in the reconstructed tract were not observed in any case of the flap technique, but were recorded in 33.3, 41.7 and 66.7%, respectively, of cases treated with McIndoe s technique. Dyspareunia was detected in 2 (9.1%) cases of the labio-vestibular technique, and in 8 (66.7%) cases of McIndoe s technique (Z14.9, pb0.01). In addition, marital failure, i.e. divorce, was recorded in 83.3% of cases in McIndoe s technique and in 22.7% of cases in the flap technique (Z and p B0.01). On the other hand, restoration of genital continuity, menstruation and fertility were not considered in any cases of McIndoe s technique. Meanwhile, continuity could be established in 6 of the 8 cases of Grade I and II Rokitanisky syndrome treated by the flap technique. Menstruation was restored in 4 cases, and fertility was restored in 3 cases, resulting in 2 full term pregnancies and 1 aborted at the 12th week. Restoration of sexuality was more promising in those cases treated by the flap technique. The mean of the scores recorded after the latter technique was significantly higher than the mean of the scores recorded after McIndoe s technique (T/df 47.32/ 32, p B0.005). Accordingly, sexuality was improved and the scores were increased by 43.9% in the cases managed by McIndoe s technique and by 110.3% in the cases managed by the labio-vestibular flap technique. Discussion Reconstruction of the vagina using the labio-vestibular flap technique is a new achievement in gynaecological surgery. This technique was actually a modification of some recent attempts in simplifying the management of Rokitansky syndrome. One of these attempts was the work of Wu et al. (8) who used the labia minora flap technique, and Sato (7) who used a vestibular flap technique in managing these cases. Sato and Wu (7,8) showed that the flap techniques were more simple, advantageous and physiological than other techniques. In addition, sexuality and sexual response were more satisfactory than the corresponding states in the other techniques, as the epithelising tissue was actually sexually sensitive genital epithelium. Accordingly, these flap techniques were theoretically considered the most suitable for managing our cases. But as most, if not all, of our patients were actually circumcised, certain modifications were needed to suit those patients. A labio-vestibular technique, not only labial or vestibular flap technique, was used. This flap was usually performed without the blind vaginal pouch being constructed non-surgically to a certain depth as most of the patients and their husbands refused that step in their treatment. In addition, it was designed without the use of tissue expansion, stent, dilatation or mould as in the original techniques. The labiovestibular flap was used to epithelise the posterior wall of the vagina and part of the lateral walls, meanwhile the anterior wall was epithelised by the original lining of the blind vaginal pouch. Comparing this technique by the commonly used McIndoe technique showed that rejection of the graft, contraction or stenosis of the neovagina, hair growth in the reconstructed tract were not seen in any case of the flap technique, unlike the cases

5 98 S.M.A. Thabet and A.H. Ali treated with McIndoe s technique. These findings may be the results of fixing the flaps in situ and its suspension to the cervix or the cervical ligaments when the latter could be identified. In addition, the flaps were prepared from non-hairy skin, so hair growth must be considered uncommon. Meanwhile, lubrication and sexuality were far better than that recorded in McIndoe s technique. The latter used the split-thickness skin graft taken from the front of the thigh. McIndoe s technique is known to be less sexually sensitive and is more liable to scarring and contraction, and usually leaves a considerable scar at its original site on the thigh. Accordingly, it is not unusual to find the gain in sexual scores after McIndoe s technique was 43.9% of the original preoperative scores; meanwhile it was 110.3% in the labio-vestibular flap technique. The latter gain in the postoperative sex scores was significantly higher than the corresponding gain in the cases treated by McIndoe s technique (T/df 47.32/32, pb0.005). This gain represents the improvement in the sexuality of both the patient and her husband as the modified sex score used involves both of them. To the best of our knowledge, no comparative sexual assessment was previously carried out between the different techniques, but some authors have recorded reports on the state of sexuality and sexual satisfaction from the point of view of the patient (10,11). The flap techniques also have the advantage of use in managing high vaginal atresia with heamatocolpos (12,13). Genital continuity was easily established by the labio-vestibular flap technique, as the flaps have to be essentially fixed to the cervix or its ligaments. Restoration of continuity was achieved in 6 cases and menstruation was achieved in 4 cases, indicating the role of the labio-vestibular flap technique in restoration of genital function. The advantage of restoration of genital continuity was not only restoration of menstruation, but also restoration of fertility. Restoration of fertility resulted in 3 pregnancies, two continuing to full term. Epithelisation of the newly formed vagina by the labio-vestibular flap may be an important factor in restoration of fertility due to its possible effect on capacitation of the sperms. Some authors with more complicated techniques of reconstruction managed similar cases of Mullerian anomaly with obstruction, but the results were less effective (14,15). Accordingly, restoration of continuity of the genital tract in cases with a functioning or hypoplastic uterus, may be one of the main advantages of the labio-vestibular technique. The flap technique was also found to be more acceptable than other techniques. The former were associated with earlier recovery and earlier establishment of sexual relations, and final results on the vulva may simulate the indistinguishable appearance of female circumcision, which is commonly seen in our patients and is accepted by them. References 1. American College of Obstetrics and Gynecology. Int J Gynecol Obstet. 2002;79(2): McIndoe AH, Banister JB. An operation for the cure of congenital absence of the vagina. J Obstet Gynecol Br. 1938;/ 45:/ Gell JS. Mullerian anomalies. Semin Reprod Med. 2003;/ 21(4):/ Williams EA. Congenital absence of the vagina, a simple operation for its relief. J Obstet Gynecol Br Commun. 1964;/ 71:/ Frank RT. The formation of an artificial vagina without operation. Am J Obstet Gynecol. 1938;/35:/ Seccia A, Salgarello M, Sturla M, Loreti A, Latorre S, Farallo E. Neovaginal reconstruction with modified McIndoe technique: a review of 32 cases. Ann Plast Surg. 2002;/49(4):/ Sato H. A new method in the treatment of vaginal agenesis with functioning uterus. Hokkaido Igaku Zasshi. 1987;/62(3):/ Wu J, Hong Y, Li SF, Hu ZQ. Labia minora skin flap vaginoplasty using tissue expansion. Zonghua Zheng Xing Wai Ke Za Zhi. 2003;/19(1):/ Thabet SMA, Thabet ASMA. Defective sexuality and female circumcision: the cause and the possible management. J Obstet Gynecol Res. 2003;/29(1):/ LeRoys. Vaginal reconstruction in adolescent females with Mayer-Rokitansky Kuster-Hauser syndrome. Plast Surg Nurs. 2001;21(1):237, Klingele CJ, Gebhart JB, Croak AJ, DiMarcdo CS, Lesnick TG, Lee RA. McIndoe procedure for vaginal agenesis: longterm outcome and effect on quality of life. Am J Obstet Gynecol. 2003;/189(6):/ Belloli G, Compobasso P, Musi L. Labial skin-flap vaginoplasty using tissue expanders. Pediatr Surg Int. 1997;/12(2/3):/ Nikolaev VV, Bizhanova DA. Perineal reconstruction in girls with high vaginal atresia. J Urol. 1998;/159(6):/ Chakravarty B, Konar H, Chowdhury RN. Pregnancies after reconstructive surgery for congenital cervicovaginal atresia. Am J Obstet Gynecol. 2000;/183:/ Creatsas G, Bakas P. A full-term pregnancy in a patient with a severe Mullerian duct anomaly with obstruction. Fertil Steril. 2000;/74(2):/4101.