Congenital penile urethrocutaneous fistula is described as an unusual developmental anomaly in children who present with an abnormal opening on the ventral aspect of penis with a normal foreskin and an absence of chordee and hypospadias. The authors present a discussion on the etiology, embryology, and management of this entity along with a description of three cases. We emphasize meticulous clinical examination for the diagnosis and to rule out other associated anomalies.
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