Congenital anterior urethrocutaneous fistula (CAUF) is a rare penile anomaly with only 63 cases reported in the literature. The anomaly can present in isolation or in association with chordee or hypospadias. We report the case of an 8-month-old boy with CAUF that resembles the embryological urethral groove. On examination, a wide urethral groove was noted to cover the midshaft of the penis with a well formed urethra extending proximally and distally and with a normal glandular anatomy, a wide glandular meatus, and a complete foreskin. The urethral groove was tubularized and covered in layers. Surgery was complicated with early superficial skin dehiscence not affecting the urethral repair.
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