Neuroleptic malignant syndrome is a serious, life threatening complication of antipsychotic therapy with typical or atypical antipsychotics (neuroleptics). It is the rarest and the most serious of neuroleptic induced movement disorders characterized by rigidity, altered cons­ciousness, fever and autonomic nervous system dysfunction. Two cases of neuroleptic malig­nant syndrome are presented. Clinical presentation, diagnostic workup, differential diagnoses, pathophysiology and treatment are discussed. Dilemmas regarding the differential diagno­sis, pathogenesis, genetic predisposition and treatment of neuroleptic malignant syndrome are addressed.