Neuroleptic malignant syndrome is a rare, but potentially life threatening, idiosyncratic reaction primarily related to use of neuroleptic medications. We report a fatal case of neuroleptic malignant syndrome in a 67 year old female with bipolar affective disorder, triggered by recent change of medications. She had marked elevation of creatinine phosphokinase levels, thrombocytopenia and acute Case reportkidney injury but no muscle rigidity. Despite early detection, intensive care support and continuous renal replacement therapy, the patient died of rhabdomyolysis induced acute kidney injury and persistent metabolic acidosis.