Published: 2016-12-14

Youssef’s Syndrome - a complication of corrective surgery for mullerian anomaly

Vineet Mishra, Suwa Saini, Rohina Aggarwal, Priyankur Roy, Shaheen Hokabaj


The presence of cervicovaginal agenesis with unicornuate uterus is a very rare mullerian anomaly. Presence of functioning unicornuate uterus poses as a great challenge for gynecologist, because a successful repair could restore normal menses and may preserve the patient’s fertility. Though, a complication of this surgery (utero-vaginal anastomosis) is Youssef’s Syndrome. We, hereby report a case of 16 year old unmarried female with complete cervicovaginal agenesis with unicornuate uterus, who initially underwent vaginoplasty (McIndoe’s) along with uterovaginal anastomosis in order to preserve her uterus, but subsequently required hysterectomy due to Youssef’s Syndrome.


Cervicovaginal agenesis, Hysterectomy, Vaginoplasty, Youssef’s Syndrome

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Heinonen PK. Uterus didelphys: a report of 26 cases. European Journal of Obstetrics and Gynecology and Reproductive Biology. 1984;17(5):345-50.

Heinonen PK. Clinical implications of the didelphic uterus: long-term follow-up of 49 cases. European Journal of Obstetrics and Gynecology and Reproductive Biology. 2000;91(2):183-90.

Abali R, Kuvat SV, Bozkurt S, Kayhan A, Yuksel MA, Caliskan H. Report of surgical correction of a cervicovaginal agenesis case: cervicovaginal reconstruction with pudendal thigh flaps. Archives of Medical Science. 2013;9(1):184-7.

Saxena, AK, Herman MI. Vaginal atresia. Webpage. Cited 02 June, 2009.

Creighton SM, Daves MC, Cutner A. Laparoscopic management of cervical agenesis. Fertil Steril. 2006;85:1510-3.

Youssef AF. Menouria following lower segment cesarean section: a syndrome. American Journal of Obstetrics and Gynecology. 1957;73(4):759-67.

Jones HW, Wheeless CR. Salvage of the reproductive potential of women with anomalous development of the mullerian ducts. American Journal of Obstetrics and Gynaecology. 1969;104:348-52.

Saraf S, Saraf P. McIndoe vaginoplasty: revisited. International Journal of Gynaecology and Obstetrics. 2007:6.

Vallerie AM, Breech L. Update in Mullerian anomalies: diagnosis, management and outcomes. Current Opinion in Obstetrics and Gynecology. 2010;22(5):381-3.

Deffanges JV, Haddad B, Muset R, Paniel BJ. Uterovaginal anastomosis in women with uterine cervix atresia - long term follow up and reproductive performance: A study of 18 cases. Hum Reprod. 2001;16:1722-5.

McIndoe A. The treatment of congenital absence and obliterative conditions of vagina. Br J Plast Surg. 1950;2(4):254-67.

Perveen K, Gupta R, Al-Badr A, Hemal AK. Robot assisted laparoscopic repair of rare post-cesarean section vesicocervical and vesicouterine fistula: a case series of a novel technique. Urology. 2012;80(2):477-82.

Narayanan P, Nobbenhuis M, Reynolds KM, Sahdev A, Reznek RH, Rockall AG. Fistulas in malignant gynaecologic disease: etiology, imaging, and management. Radiographics. 2009;29(4):1073-83.