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We describe a case of posterior urethral valve with multiple urethroperineal fistulas. This association is extremely rare. The crucial point in the diagnosis is to distinguish these fistulas from the urethral duplications that actually guide the treatment. The presence of these fistulas in patients with posterior urethral valve may have a beneficial effect on renal function as it reduce the pressure on the bladder. On the other hand, incomplete valve ablation may contribute to the recurrence of the fistula.

Multiple urethroperineal fistula in a boy with posterior urethral valve: A case report of an extremely rare association