Bicornuate uterine horns with complete cervical–vaginal agenesis and congenital vesicouterine fistula

Abstract

We present a case of bicornuate uterine horns with complete cervico-vaginal agenesis and associated congenital uterovaginal fistula. The patient presented with cyclical hematuria, amenorrhea, and abdominal lump. The vagina was blind-ending. The cystoscopic examination during cyclical hematuria revealed bloody efflux through a small fistula on the posterior wall of the urinary bladder. The magnetic resonance imaging (MRI) showed bicornuate uterus and cervical agenesis. Hysterectomy and repair of the uterovesical fistula was done. The vagina was reconstructed using an amniotic mould. The report underlies the importance of MRI in diagnosing complexity of such rare anomalies. It also stresses for the need of hysterectomy if cervicovaginal agenesis is present.

Congenital vesicouterine fistulas—A PRISMA‐compliant systematic review

“…Besides the Williams et al case where vaginal inspection in a 9‐month infant was not reported, all others were with vaginal atresia or agenesis. Seven of nine (77.78%) cases included distal vaginal atresia in their picture, whereas two patients described by Daskalov and Šahpazov and by Kumar et al slightly differed, demonstrating atresia of the upper third of the vagina and of the cervix, and complete cervico‐vaginal agenesis, respectively. Thus, the level of vaginal outlet obstruction may be secondary to the primary factor of the reproductive tract's lack of patency, which seems to be a prerequisite for the formation of the congenital fistula.…”

“…Figure presents the flowchart for the identification of relevant literature sources. Ten studies were identified, all singular cases (Table ) . One reference not covered by PubMed ® was obtained from the ResearchGate networking service as feedback information sent to a first author .…”

“…Ten studies were identified, all singular cases (Table 1). [23][24][25][26][27][28][29][30][31][32] One reference not covered by PubMed ® was obtained from the ResearchGate networking service as feedback information sent to a first author. 32 In contrast, a report describing a case of vesicocervical fistula with vaginal atresia, uterus unicornis, absent left adnexa, and left kidney agenesis in a 21-year old was not included because it was not apparent from the description that it was a congenital defect since her menouria manifested as late as 3 years after menarche.…”

Iatrogenic urethrovaginal fistula with transverse vaginal septum presenting as cyclical hematuria

“…Congenital vesicouterine, cervicovesical, and vesicovaginal fistula associated with other mullarian anomalies has also been reported in literature. [4–6] Iatrogenic fistulous communication between the genital tract and the urinary tract, known to occur as a complication of cesarean section, has been described in literature as “Youssef's syndrome.” The uterine side of the fistulous tract is above the uterine isthmus. It is characterized by cyclical hematuria, absence of vaginal bleeding, and complete urinary continence.…”

(Ventrodorsal) Symmetrical Bicornuate Uterus Mimicking a Pedunculated Myoma—A Case Report

“…Kumar et al (2008) [4] in a study reported that MRI showing bicornuate uterus exposed cervical agenesis. Some studies [5] have cited septate uterus as the 2 nd most common uterine anomaly after bicornuate uterus [6].…”