Background <p>Persistent urogenital sinus is a rare (6/100,000) cloacal anomaly in which the urinary and genital tracts do not differentiate during embryonic development due to intrauterine androgen exposure, anorectal malformation, or an isolated form independent of both.</p> Case Report <p>A 20&#xa0;year old nulligravida presented with cyclical hematuria during menstruation and inability to have penetrative coitus. She attained menarche at 14&#xa0;years of age and had normal menstrual cycles. Her general and systemic examination were normal. Examination of external genitalia revealed normal clitoris with an external urethral meatus; fused labia minora; normal labia majora; blind vaginal dimple; deficient perineum and normal anal orifice. MRI pelvis showed bicornuate unicollis uterus, normal ovaries, hydrocolpos, patent vagina with atresia in distal most part and normal renal system. Examination under anaesthesia was done, bladder catheterised and labia minora separated by sharp dissection. During the process of vaginal dilatation, Foleys bulb was felt. On suspicion of bladder injury, cystoscopy was done which revealed an intact bladder. Urethroscopy done revealed a common tract communicating with bladder neck anteriorly and vagina posteriorly. 2 urethral orifices were identified- 1 in the clitoris, another continent meatus opening into the common tract. Vaginal mucosa was brought down upto introitus and sutured. Postoperative vaginal dilatation was done and endometrium suppressed with progesterones. Patient voided urine normally after removal of catheter and is under follow up.</p> Conclusion <p>A high index of suspicion, meticulous examination, imaging modalities, multidisciplinary approach and thorough knowledge of anatomy are essential for diagnosis and repair of PUGS.</p>