Alaa Cheikhelard1, Maud Bidet2, Amandine Baptiste3, Magali Viaud2, Christine Fagot4, Naziha Khen-Dunlop5, Christine Louis-Sylvestre6, Sabine Sarnacki5, Philippe Touraine7, Caroline Elie3, Yves Aigrain8, Michel Polak9. 1. Departments of Pediatric Surgery and Urology, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France; National Reference Center for Rare Gynecological Diseases (PGR), Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France. Electronic address: alaa.cheikhelard@aphp.fr. 2. Department of Pediatric Endocrinology, Gynecology, and Diabetology, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France; National Reference Center for Rare Gynecological Diseases (PGR), Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France. 3. Clinical Research Unit/Clinical Investigation Center, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France. 4. Department of Pediatric Endocrinology, Gynecology, and Diabetology, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France. 5. Departments of Pediatric Surgery and Urology, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France; Université Paris Descartes, Paris, France. 6. Department of Obstetrics and Gynecology, Institut Mutualiste Montsouris, Paris, France. 7. Department of Endocrinology and Reproductive Medicine, Assistance Publique - Hôpitaux de Paris, Institut Endocrinologie, Médecine Interne et Maladies Métaboliques, Hôpital Pitié-Salpêtrière, Paris, France; Médecine Sorbonne Université, Paris, France. 8. Departments of Pediatric Surgery and Urology, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France; National Reference Center for Rare Gynecological Diseases (PGR), Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France; Université Paris Descartes, Paris, France. 9. Department of Pediatric Endocrinology, Gynecology, and Diabetology, Assistance Publique - Hôpitaux de Paris, Hôpital Necker, Paris, France; Université Paris Descartes, Paris, France.
Abstract
BACKGROUND: Vaginal agenesis in Mayer-Rokitansky-Küster-Hauser syndrome can be managed either by various surgeries or dilation. The choice still depends on surgeon's preferences rather than on quality comparative studies and validated protocols. OBJECTIVE: We sought to compare dilation and surgical management of vaginal agenesis in Mayer-Rokitansky-Küster-Hauser syndrome, in terms of quality of life, anatomical results, and complications in a large multicenter population. STUDY DESIGN: Our multicenter study included 131 patients >18 years, at least 1 year after completing vaginal agenesis management. All had an independent gynecological evaluation including a standardized pelvic exam, and completed the World Health Organization Quality of Life instrument (general quality of life) as well as the Female Sexual Function Index and Female Sexual Distress Scale-Revised (sexual quality of life) scales. Groups were: surgery (N = 84), dilation therapy (N = 26), and intercourse (N = 20). One patient was secondarily excluded because of incomplete surgical data. For statistics, data were compared using analysis of variance, Student, Kruskal-Wallis, Wilcoxon, and Student exact test. RESULTS: Mean age was 26.5 ± 5.5 years at inclusion. In all groups, World Health Organization Quality of Life scores were not different between patients and the general population except for lower psychosocial health and social relationship scores (which were not different between groups). Global Female Sexual Function Index scores were significantly lower in the surgery and dilation therapy groups (median 26 range [2.8-34.8] and 24.7 [2.6-34.4], respectively) than the intercourse group (30.2 [7.8-34.8], P = .044), which had a higher score only in the satisfaction dimension (P = .004). However, the scores in the other dimensions of Female Sexual Function Index were not different between groups. The Female Sexual Distress Scale-Revised median scores were, respectively, 17 [0-52], 20 [0-47], and 10 [10-40] in the surgery, dilation therapy, and intercourse groups (P = .38), with sexual distress in 71% of patients. Median vaginal depth was shorter in dilatation therapy group (9.6 cm [5.5-12]) compared to surgery group (11 cm [6-15]) and intercourse group (11 cm [6-12.5]) (P = .039), but remained within normal ranges. One bias in the surgery group was the high number of sigmoid vaginoplasties (57/84, 68%), but no differences were observed between surgeries. Only 4 patients achieved vaginas <6.5 cm. Delay between management and first intercourse was 6 months (not significant). Seventy patients (53%) had dyspareunia (not significant), and 17 patients all from the surgery group had an abnormal pelvic exam. In the surgery group, 34 patients (40.5%) had complications, requiring 20 secondary surgeries in 17 patients, and 35 (42%) needed postoperative dilation. In the dilation therapy group, 13 (50%) needed maintenance dilation. CONCLUSION: Surgery is not superior to therapeutic or intercourse dilation, bears complications, and should therefore be only a second-line treatment. Psychological counseling is mandatory at diagnosis and during therapeutic management.
BACKGROUND: Vaginal agenesis in Mayer-Rokitansky-Küster-Hauser syndrome can be managed either by various surgeries or dilation. The choice still depends on surgeon's preferences rather than on quality comparative studies and validated protocols. OBJECTIVE: We sought to compare dilation and surgical management of vaginal agenesis in Mayer-Rokitansky-Küster-Hauser syndrome, in terms of quality of life, anatomical results, and complications in a large multicenter population. STUDY DESIGN: Our multicenter study included 131 patients >18 years, at least 1 year after completing vaginal agenesis management. All had an independent gynecological evaluation including a standardized pelvic exam, and completed the World Health Organization Quality of Life instrument (general quality of life) as well as the Female Sexual Function Index and Female Sexual Distress Scale-Revised (sexual quality of life) scales. Groups were: surgery (N = 84), dilation therapy (N = 26), and intercourse (N = 20). One patient was secondarily excluded because of incomplete surgical data. For statistics, data were compared using analysis of variance, Student, Kruskal-Wallis, Wilcoxon, and Student exact test. RESULTS: Mean age was 26.5 ± 5.5 years at inclusion. In all groups, World Health Organization Quality of Life scores were not different between patients and the general population except for lower psychosocial health and social relationship scores (which were not different between groups). Global Female Sexual Function Index scores were significantly lower in the surgery and dilation therapy groups (median 26 range [2.8-34.8] and 24.7 [2.6-34.4], respectively) than the intercourse group (30.2 [7.8-34.8], P = .044), which had a higher score only in the satisfaction dimension (P = .004). However, the scores in the other dimensions of Female Sexual Function Index were not different between groups. The Female Sexual Distress Scale-Revised median scores were, respectively, 17 [0-52], 20 [0-47], and 10 [10-40] in the surgery, dilation therapy, and intercourse groups (P = .38), with sexual distress in 71% of patients. Median vaginal depth was shorter in dilatation therapy group (9.6 cm [5.5-12]) compared to surgery group (11 cm [6-15]) and intercourse group (11 cm [6-12.5]) (P = .039), but remained within normal ranges. One bias in the surgery group was the high number of sigmoid vaginoplasties (57/84, 68%), but no differences were observed between surgeries. Only 4 patients achieved vaginas <6.5 cm. Delay between management and first intercourse was 6 months (not significant). Seventy patients (53%) had dyspareunia (not significant), and 17 patients all from the surgery group had an abnormal pelvic exam. In the surgery group, 34 patients (40.5%) had complications, requiring 20 secondary surgeries in 17 patients, and 35 (42%) needed postoperative dilation. In the dilation therapy group, 13 (50%) needed maintenance dilation. CONCLUSION: Surgery is not superior to therapeutic or intercourse dilation, bears complications, and should therefore be only a second-line treatment. Psychological counseling is mandatory at diagnosis and during therapeutic management.
Keywords:
Female Sexual Distress Scale-Revised; Female Sexual Function Index; Mayer-Rokitansky-Küster-Hauser syndrome; World Health Organization Quality of Life instrument; multicenter study; quality-of-life studies; sexual distress; vaginal aplasia; vaginal dilation; vaginoplasty