Background: A 16-year-old boy with penilurethra anomaly was admitted to our clinic. This is a case description of a hypospadiac congenital urethrocutaneous fistula of the anterior urethra. Methods/Results: After the chordee was corrected by removing dysgenetic tissue in urethral plate, urethral defect was repaired with tranverse preputial island flap. Conclusions: Congenital urethrocutaneous fistula is rare. Thirty-two cases of urethrocutaneous fistula have been reported previously. Embryological events that cause anterior urethrocutaneous fistula are unclear. We reported a case with hypospadiac congenital urethrocutaneous fistula of the anterior urethra treated with tranverse preputial island flap.