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Urethral duplications are rare lower urinary tract anomalies. Here we present an unusual presentation of urethral duplication in which, completely patent accessory urethra was coursing laterally on perianal region, near the anus, and presented as perianal fistula. A 2-month-old boy presented with discharge from the perianal region. On physical examination a fistula was observed on perianal region, in the 1 o’clock position and only 1 cm near the anus. Fistulography showed an opening to the prostatic urethra. The opening to the urethra was seen left superior part of the verumontanum on cystourethroscopy by giving methylene blue from the fistula.  The accessory urethra was dissected from the skin to the prostatic part of the orthotopic urethra and excised completely with anterior sagittal approach. His postoperative course was uneventful.  In unusual form of urethral duplications of Effmann Type IIA2, as in our case, orthotopic urethra is normal and ventral urethra opens to the perineum. It should be kept in mind that especially laterally placed ones can be misdiagnosed as anal fistula.

Y-Type Urethral Duplication Presented with Perianal Fistula in an Infant