Neomycin is known for its ototoxicity after many different methods of administration of the drug. Neomycin sometimes is used for bladder irrigation in patients with urinary tract infections when other treatments fail. To our knowledge toxicity after bladder instillation of neomycin has not been reported previously. We report a case of perception deafness in a boy with end stage renal disease who underwent neomycin bladder instillations following augmentation of the bladder with a megaureter.