A 3-year-old boy presented to our pediatric urology with a history of urine flow under the scrotum when voiding in a squatting position but not when standing. And the ventral side of the front penis became enlarged during urination and dribbled afterward. Physical examination revealed the boy had 2 urethras opening at the tip of glans, and another accessory urethra opening at perineum. Rigid cystoscopy and voiding cystourethrography confirmed it to be a urethral triplication malformation. This condition, the combination of urethral diverticulum and urethral triplication, consisting of urethro-perineum fistula, has not been previously reported.
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