We report 2 cases of persistent urogenital sinus (UGS) with duplication of the bladder and vagina in different planes. Cystoscopy and vaginoscopy were used to diagnose persistent UGS with bladder and vaginal duplication in 2 infants with recurrent urinary tract infection. Surgical repair was done by way of a midline abdominal and perineal approach using tubularized UGS to reconstruct the urethra. Persistent UGS represents a major anomaly; these patients had the added complexity of duplication of the bladder and vagina. In these complex patients, the true anatomy may only become apparent during the surgical reconstruction, necessitating a flexible, individual approach.
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