To present the case of a 5-day-old male infant referred to our clinic with complaints of huge swollen testes, recurrent urinary tract infection, and diarrhea. The imaging studies and surgical assessments revealed a urethrorectal fistula and 2 nonfunctional urethras. Cutaneous vesicostomy was performed urgently to avoid additional renal infection. At the age of 6 months, the anterior anal insertion was repaired by perineal access. Eventually, urethral reconstruction was performed when the boy was 3 years old. The patient was asymptomatic at the last follow-up examination without additional urinary tract infections. The combination of urethrovasal reflux and congenital urethral triplication, consisting of urethrorectal fistula, has not been previously reported.
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