Aripiprazole, an atypical antipsychotic with a novel method of action, has only recently been awarded a license in the UK. We report our first patient to receive this drug, who had treatment-resistant schizophrenia and developed neuroleptic malignant syndrome (NMS) with aripiprazole. To our knowledge, this is the first published case report involving aripiprazole and NMS in a potentially fatal medical emergency. Further experience with this drug should indicate whether this is an isolated case (as described with other atypical antipsychotics) or constitutes a more serious risk than that suggested by the relatively beneficial therapeutic profile described in the literature to date.