Abstract
This case report aimed to describe the clinical presentation and surgical management of a congenital anterior urethral diverticulum (CAUD) with parental consanguinity. A 4-year-old boy presented to our center, complaining of increasingly difficult urination for two days. His parents were Saudi cousins. A clean catch urine sample was positive for E. coli and was sensitive to Cefuroxime. A voiding cystourethrogram revealed a penile anterior urethral diverticulum. The penis was degloved, the cavity of the anterior urethral diverticulum was opened, the edges were excised, and the mouth of the diverticulum was closed. Consanguinity as a risk factor remains to be explored.