Abstract Single system ectopic ureters are rare and bilateral single system ectopic ureters (BSSEU) are even rarer. Common locations of openings of such ureters include vagina and vestibule in the females, and posterior urethra and seminal vesicles in the males. BSSEU opening into a vaginalized urogenital sinus with absence of urinary bladder is an extremely rare occurrence. Such a case is being reported and its management is discussed briefly.