Background: A 3-year-old boy visited our hospital for aberrance of urination. He had a fistula on his ventral penile shaft. Our diagnosis was congenital urethrocutaneous fistula.
Methods/results: We performed one-stage repair transverse preputial onlay island flap urethroplasty. Postoperatively, the patient was voiding comfortably with no recurrence of fistula.
Conclusions: Congenital urethrocutaneous fistula is rare. Eighteen cases of congenital urethrocutaneous fistula have been reported previously. We consider the etiology of congenital urethrocutaneous fistula as a deficiency of the urethral plate and fusion of urethral folds.