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Parkinsonism- hyperpyrexia syndrome (PHS) is a neurological emergency that mimics neuroleptic malignant syndrome and sepsis. Abrupt cessation of anti parkinsons drugs, usually levodopa is responsible for this syndrome. Relative dopamine deficiency is proposed mechanism for PHS and replacement of dopaminergic drugs is the mainstay of treatment. We report a case who presented with manifestations of PHS after missing a single dose of levodopa.