Rhabdomyosarcoma of the genitourinary tract is rare and predominantly affects paediatric patients. We present an unusual case of such a lesion in an adult with extensive occupation of the bladder cavity by the lesion, resulting in bilateral ureteric obstruction, without evidence of ureteric outlet invasion. We outline the unusual CT and macroscopic appearance of this lesion. We also discuss the literature data and management strategies of rhabdomyosarcoma of the genitourinary tract.