Unusual Case of Urethrorectal Fistula in Adolescence in a Patient with a History of Congenital Anorectal Malformation

BACKGROUND Urethrorectal fistula is a rare and debilitating condition. Spontaneous closure is rarely effective, and appropriate management regarding timing of repair and surgical approach remains controversial. CASE PRESENTATION We present a case of an 18-year-old male found to have a urethrorectal fistula after diagnostic work up for unejaculation. The patient gradually developed recurrent urinary tract infections and urine and semen leak from his rectum. He had a medical history of an anorectal reconstruction in the second postnatal day due to an anorectal malformation. Imaging with a rectal endoscopic ultrasound scan revealed a suprasphincteric urethrorectal fistula that was further confirmed with semirigid urethrocystoscopy and placement of a nitinol guidewire through the urethral fistula orifice. Its anal orifice was 3 cm above the anal verge at the 12th hour of the rectum. The fistula orifice on the rectum was identified with a transanal approach and the fistula was managed with the performance of an advancement mucosal flap and bladder catheterization. The patient developed a recurrence with this approach and finally underwent fistula ligation and reconstruction using a scrotal flap. The patient has not had a recurrence of the fistula during his follow-up. CONCLUSION This is an unusual case of iatrogenic urethrorectal fistula as it presented in adolescence many years from the initial operation of anorectal reconstruction and with unusual symptoms.