Congenital anterior urethrocutaneous fistula (CAUF) is an extremely rare penile anomaly which presents despite a normal urethral meatus and intact prepuce. It may present in isolation or with associated anomalies such as hypospadias or chordee. We report the case of a 14-months-old boy with a congenital anterior urethrocutaneous fistula on the ventral penile shaft. A single-stage urethrocutaneous fistula closure with a dartos flap along with torsion correction was performed. The postoperative course was uneventful, and the child remains well on follow-up.
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