Female hypospadias is a rare malformation. We report on an unusual case that presented with a urethral dimple in the perineum in association with a pelvic soft tissue sarcoma compressing and displacing the urethra. The diagnosis was made by cystoscopy, and, as the patients remained asymptomatic, no treatment was eventually required for her urologic malformation. This is, to our knowledge, the first reported case of female hypospadias associated with a perineal urethral dimple. The possible pathogenesis of the malformation is discussed also in relation to the simultaneous presence of a pelvic mass.