Variants of congenital prepubic sinus have been reported rarely, and because the anatomic features often differ from each other, a consensus concerning the embryology and classification was not achieved yet. Various names including congenital prepubic sinus, sub pubic fistula, prepubic dermoid sinus, and suprapubic dermoid sinus, were used to identify these lesions, and, among the classifications available, none seems to clearly describe this entity. We present a 2-year old boy with a case of epispadiac variant of urethral duplication in which the duplicated urethra presented as a prepubic sinus. We report this uncommon anomaly and review the scattered published reports to improve the global understanding of this uncommon congenital lesion.