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Complete Adult Vulvar Fusion: A Case Report
COMPLETE ADULT VULVAR FUSION: A CASE REPORT Bernard Lambert, MD, FRCSC Departement d'obstetrique gynecologie, Centre hospitalier universitaire de Montreal, Montreal QC
Abstract Background: Labial fusion is most commonly seen in prepubertal girls and in postmenopausal women affected by advanced lichen sclerosus et atrophicus. Postpartum labial fusion is a rare phenomenon for a woman with no evidence of hypoestrogen ism. Case: A woman with a normal delivery and a normal postpartum examination presented 7 years later with a complete vulvar closure from the urethral opening to the posterior fourchette. Conclusion: Complete vulvar fusion can rarely occur without any evidence of hypoestrogenism. It is managed by surgical incision and perineorrhaphic correction without sequelae.
may also occur in prepubescent girls, It has also been observed in advarIced lichen sclerosus et atrophicus. 5 Labial fusion could be compared to male phimosis, 1 and advanced treatment is otten limited. 6 Corroy et al. 2 reported two cases oflabial synechiae in a 74year-old postmenopausal woman and an 88-year-old postmenopausal womarI, likely related to hypoestrogenism arId local atrophy, similar to the cases found in infants. Both women experienced dysuria arId vaginal urinary retention, following intravaginal micturition.
Resume Contexte : La fusion labiale est Ie plus souvent constatee chez les filles prepubertaires et les femmes menopausees qui sont atteintes d'une forme avancee de lichen sclereux et atrophique. La fusion labiale post-partum est un phenomEme rare chez les femmes qui ne presentent aucun signe d'hypo-
THE CASE
~strogenie.
Cas : Une femme ayant vecu un accouchement normal et obtenu des resultats normaux a la suite de I'examen postpartum a presente, sept ans plus tard, une occlusion vulvaire complete (du meat uretral a la fourchette posterieure). Conclusion : La fusion vulvaire complete peut rarement se produire en I'absence de signes d'hypo-~strogEmie. Elle est prise en charge par incision chirurgicale et reparation perineorraphique, et ce, sans sequelles.
J Obstet Gynaecol
Can 2004;26(5):501-2.
INTRODUCTION
Labial fusion is a rare phenomenon. 1-3 A 1% incidence oflabial fusion has been reported in girls under 2 years of age,4 but it
KeyWords Membrane fusion; genitalia, female, abnormalities; vulvar abnormalities Competing interests: None declared. Received on July 28, 2003 Revised and accepted on October 14, 2003 IOGC
SUSarI (pseudonym), a 42-year-old womarI, presented in February 2002 with complaints of being unable to have sexual intercourse since her second vaginal delivery 7 years earlier. SUSarI described her previous sexual activity as having been normal, although with occasional pain and difficulty with vaginal orgasm. SUSarI gave birth spontarIeously under pudendal block. She suffered a third-degree laceration, which was identified, arId the arIal sphincter arId perineal elevator muscles were repaired without arIy reported difficulty. There was no postpartum hemorrhage, arId no infection occurred in the immediate postpartum period. SUSarI discontinued breastfeeding after a few days of nursing because of insufficient milk production. There was no evidence of mammary congestion. Susan's 6-week postpartum examination was normal, and a normal vaginal opening was observed. No internal vaginal examination was performed at that time or for 7 years. From that time, Susan had normal menstrual cycles every 28 days with 4 to 5 days' duration of flow. Sexual activity was limited to vulvar arId clitoral stimulation. When Susan finally brought the sexual problem to her physician's attention, vulvar inspection revealed a complete vulvar fusion extending arIteriorly to the urethral meatus with complete vaginal closure. No hormonal investigation occurred as Susan's menstrual cycles were regular. Similarly, no attempt was made to use topical hormones. Rather, SUSarI was admitted MAY 2004
for surgical correction of the vulvar fusion. Under general anaesthesia, her vagina was opened from I cm under the urethral orifice with a grooved canula, to approximately 3 cm from the posterior fourchette. The posterior vaginal mucosa was undermined, pulled down, and sutured to the vulva. Dexon 2-0 sutures were used to approximate the incision sides. Susan's postoperative course was uneventful. At her I-month and 3-month postoperative follow-ups, Susan's introitus was observed to be healed. Susan reported that vaginal intercourse was now possible, and her feeling during sexual intercourse and orgasm had returned to her normal condition. Susan was encouraged to keep up regular sexual activity so as to avoid reccurrence of fusion. Oral contraception was prescribed.
ACKNOWLEDGEMENTS The woman whose story is told in this case report has provided signed permission for its puplication. REFERENCES I. Kuo DM. Chuang CK. Hsieh CC. Liou JD. Chen KC. Hsieh TT. Labial fusion in a thirty-year-old woman. Acta Obstet Gynecol Scand
1998;77(6):697-8. 2. Corroy JS. Girot V. Amicabile C. Guillemin P. Synechiae of the labia minora as a cause of dysuria. Ann Urol (Paris) 1989;23(6):504-5. 3. Uei 1; Katou Y, Shimizu N.Yamanaka H. Seki M.lbuki R. Labial adhesion in a reproductive woman with difficulties of sexual intercourse and urination. Hinyokika Kiyo 2000;46(6):433-6. 4. EvrUke C.Ozgiinen F1; Kadayif~i O. Atay Y, Demir C. Aridogan N. Labial fusion in a pubertal girl: a case report.J Pediatr Adolesc Gynecol
DISCUSSION
Complete vulvar fusion postpartum is a rare phenomenon? and its pathogenesis is unknown. 8 Hypoestrogenism in childhood9 and in elderly women2 can explain spontaneous labial fusion. This condition is normally treated with local estrogen cream. Although hypoestrogenism may contribute to stricture of the introitus in postmenopausal women? usually vulvar pathology, such as lichen sclerosus et atrophicus, is observed. In this situation, progressive narrowing of the introitus first limits vaginal intercourse, and in later stages, may interfere with micturition and urinary flow. Susan developed complete vaginal closure, with normal vulvar teguments and no evidence oflichen sclerosus or hypoestrogenism. 1,8
JOGC
1996;9:81-2. 5. Neill SM. Tatnall FM. Cox NH. Guidelines for the management of lichen sclerosus. Br J Dermatol 2002; 147:640-9. 6. Kartamaa M. Reitamo S. Treatment of lichen sclerosus with carbon dioxide laser vaporization. Br J Dermatol 1997; 136:356-9. 7. Brisson P, Patel H. Feins N. Female circumcision.J Pediatr Surg
200 I;36(7): I068-9. 8. Arkin AE. Chern-Hughes B. Case report labial fusion postpartum and clinical management of labiallacerations.J MidwiferyWomens Health
2002;47(4):290-2. 9. Omar HA. Management of labial adhesions in prepubertal girls.J Pediatr Adolesc Gynecol 2000; 13(4): 183-5.
MAY 2004
Abstract Background: Labial fusion is most commonly seen in prepubertal girls and in postmenopausal women affected by advanced lichen sclerosus et atrophicus. Postpartum labial fusion is a rare phenomenon for a woman with no evidence of hypoestrogen ism. Case: A woman with a normal delivery and a normal postpartum examination presented 7 years later with a complete vulvar closure from the urethral opening to the posterior fourchette. Conclusion: Complete vulvar fusion can rarely occur without any evidence of hypoestrogenism. It is managed by surgical incision and perineorrhaphic correction without sequelae.
may also occur in prepubescent girls, It has also been observed in advarIced lichen sclerosus et atrophicus. 5 Labial fusion could be compared to male phimosis, 1 and advanced treatment is otten limited. 6 Corroy et al. 2 reported two cases oflabial synechiae in a 74year-old postmenopausal woman and an 88-year-old postmenopausal womarI, likely related to hypoestrogenism arId local atrophy, similar to the cases found in infants. Both women experienced dysuria arId vaginal urinary retention, following intravaginal micturition.
Resume Contexte : La fusion labiale est Ie plus souvent constatee chez les filles prepubertaires et les femmes menopausees qui sont atteintes d'une forme avancee de lichen sclereux et atrophique. La fusion labiale post-partum est un phenomEme rare chez les femmes qui ne presentent aucun signe d'hypo-
THE CASE
~strogenie.
Cas : Une femme ayant vecu un accouchement normal et obtenu des resultats normaux a la suite de I'examen postpartum a presente, sept ans plus tard, une occlusion vulvaire complete (du meat uretral a la fourchette posterieure). Conclusion : La fusion vulvaire complete peut rarement se produire en I'absence de signes d'hypo-~strogEmie. Elle est prise en charge par incision chirurgicale et reparation perineorraphique, et ce, sans sequelles.
J Obstet Gynaecol
Can 2004;26(5):501-2.
INTRODUCTION
Labial fusion is a rare phenomenon. 1-3 A 1% incidence oflabial fusion has been reported in girls under 2 years of age,4 but it
KeyWords Membrane fusion; genitalia, female, abnormalities; vulvar abnormalities Competing interests: None declared. Received on July 28, 2003 Revised and accepted on October 14, 2003 IOGC
SUSarI (pseudonym), a 42-year-old womarI, presented in February 2002 with complaints of being unable to have sexual intercourse since her second vaginal delivery 7 years earlier. SUSarI described her previous sexual activity as having been normal, although with occasional pain and difficulty with vaginal orgasm. SUSarI gave birth spontarIeously under pudendal block. She suffered a third-degree laceration, which was identified, arId the arIal sphincter arId perineal elevator muscles were repaired without arIy reported difficulty. There was no postpartum hemorrhage, arId no infection occurred in the immediate postpartum period. SUSarI discontinued breastfeeding after a few days of nursing because of insufficient milk production. There was no evidence of mammary congestion. Susan's 6-week postpartum examination was normal, and a normal vaginal opening was observed. No internal vaginal examination was performed at that time or for 7 years. From that time, Susan had normal menstrual cycles every 28 days with 4 to 5 days' duration of flow. Sexual activity was limited to vulvar arId clitoral stimulation. When Susan finally brought the sexual problem to her physician's attention, vulvar inspection revealed a complete vulvar fusion extending arIteriorly to the urethral meatus with complete vaginal closure. No hormonal investigation occurred as Susan's menstrual cycles were regular. Similarly, no attempt was made to use topical hormones. Rather, SUSarI was admitted MAY 2004
for surgical correction of the vulvar fusion. Under general anaesthesia, her vagina was opened from I cm under the urethral orifice with a grooved canula, to approximately 3 cm from the posterior fourchette. The posterior vaginal mucosa was undermined, pulled down, and sutured to the vulva. Dexon 2-0 sutures were used to approximate the incision sides. Susan's postoperative course was uneventful. At her I-month and 3-month postoperative follow-ups, Susan's introitus was observed to be healed. Susan reported that vaginal intercourse was now possible, and her feeling during sexual intercourse and orgasm had returned to her normal condition. Susan was encouraged to keep up regular sexual activity so as to avoid reccurrence of fusion. Oral contraception was prescribed.
ACKNOWLEDGEMENTS The woman whose story is told in this case report has provided signed permission for its puplication. REFERENCES I. Kuo DM. Chuang CK. Hsieh CC. Liou JD. Chen KC. Hsieh TT. Labial fusion in a thirty-year-old woman. Acta Obstet Gynecol Scand
1998;77(6):697-8. 2. Corroy JS. Girot V. Amicabile C. Guillemin P. Synechiae of the labia minora as a cause of dysuria. Ann Urol (Paris) 1989;23(6):504-5. 3. Uei 1; Katou Y, Shimizu N.Yamanaka H. Seki M.lbuki R. Labial adhesion in a reproductive woman with difficulties of sexual intercourse and urination. Hinyokika Kiyo 2000;46(6):433-6. 4. EvrUke C.Ozgiinen F1; Kadayif~i O. Atay Y, Demir C. Aridogan N. Labial fusion in a pubertal girl: a case report.J Pediatr Adolesc Gynecol
DISCUSSION
Complete vulvar fusion postpartum is a rare phenomenon? and its pathogenesis is unknown. 8 Hypoestrogenism in childhood9 and in elderly women2 can explain spontaneous labial fusion. This condition is normally treated with local estrogen cream. Although hypoestrogenism may contribute to stricture of the introitus in postmenopausal women? usually vulvar pathology, such as lichen sclerosus et atrophicus, is observed. In this situation, progressive narrowing of the introitus first limits vaginal intercourse, and in later stages, may interfere with micturition and urinary flow. Susan developed complete vaginal closure, with normal vulvar teguments and no evidence oflichen sclerosus or hypoestrogenism. 1,8
JOGC
1996;9:81-2. 5. Neill SM. Tatnall FM. Cox NH. Guidelines for the management of lichen sclerosus. Br J Dermatol 2002; 147:640-9. 6. Kartamaa M. Reitamo S. Treatment of lichen sclerosus with carbon dioxide laser vaporization. Br J Dermatol 1997; 136:356-9. 7. Brisson P, Patel H. Feins N. Female circumcision.J Pediatr Surg
200 I;36(7): I068-9. 8. Arkin AE. Chern-Hughes B. Case report labial fusion postpartum and clinical management of labiallacerations.J MidwiferyWomens Health
2002;47(4):290-2. 9. Omar HA. Management of labial adhesions in prepubertal girls.J Pediatr Adolesc Gynecol 2000; 13(4): 183-5.
MAY 2004