Congenital anterior urethrocutaneous fistula is an extremely rare anomaly which may present in an isolated form or accompanying hypospadias or chordee. We describe an 8-month-old boy who presented to our outpatient clinic with a congenital anterior urethrocutaneous fistula on the ventral aspect of the penile shaft. He underwent single stage tubularized incised plateurethroplasty with a dartos flap cover. Postoperatively, the patient recovered without any complications and is doing fine on the follow-up visits.
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