The neuroleptic malignant syndrome is a rare and potentially lethal reaction which is usually associated with the use of medications with antidopaminergic properties. This article describes the case of an elderly gentleman who developed the neuroleptic malignant syndrome after withdrawal of the anticholinergic agent benzhexol (trihexyphenidyl hydrochloride), which he was taking for Parkinson’s disease. The patient improved rapidly after treatment was restarted. This case adds to the evidence that antidopaminergic agents may not be necessary for the development of this syndrome, and increased awareness of this possibility is advisable in such circumstances.peer-reviewe