Neuroleptic malignant sydnrome (NMS) is a rare but potentially life-threatening idiosyncratic adverse reaction, usually seen with antipsychotic treatment. It is characterized by fever, autonomic instability, extrapyramidal syndrome and altered mental state. Raised serum creatine kinase level, impaired liver and kidney function tests, leucocytosis and electrolyte disturbance may be the accompanying laboratory features. Various demographic, patient-medication and environmental factors increase the risk of NMS. In this case report, the clinical characteristics and management of NMS in a patient who has several risk factors such as mental retardation, use of parenteral, high dose typical antipsychotic combination for long duration is discussed.