We report on 3 patients with a midline sinus extending from the skin overlying the pubis to the anterior wall of the bladder. Two sinuses were lined by transitional epithelium. The sinuses were surrounded by bundles of smooth muscle and lacked any inflammatory reaction, which suggests a developmental origin. Possible etiological causes for this unusual congenital anomaly are discussed. We conclude that these sinuses may represent a variant of epispadiac duplication of the urethra.