Currently there are scanty data in the literature on outcome of kidney transplantation in mentally retarded patients. In subjects with significant mental retardation, co-operative patient supervised by a reliable long term caregiver with long life expectancy and able to take medication under supervision, are only selected for transplantation till today. A 30yr old male known to have cerebral palsy with severe mental retardation [I Q < 30] and on antiepileptic drugs since the age of 8 months underwent renal transplantation with ATG induction and triple drug maintenance immunosuppression. Native kidney disease was bilateral multicystic kidneys and was on hemodialysis for few weeks prior to renal transplant. Patient was restless and non compliant to dialysis. There was immediate diuresis post transplant with excellent graft function and patient received standard fluid management during and after surgery. He was sedated and restrained during initial post operative days for better care and management. Later he was off sedation and compliance to immunosuppressive and other supportive treatment was excellent. Renal transplantation is a viable treatment in ESRD patients with mental retardation provided a supportive long term care giver is available.