We recently observed a 45-year-old patient with a history of psychiatric illness who presented with severe hyperthermia (rectal temperature above 41 degrees C) with intense rhabdomyolysis and liver cytolysis during tetrabenazine therapy for neuroleptic tardive dyskinesia. In addition to tetrabenazine, this patient took lorazepam and two antidepressant drugs: clomipramine, a potent serotonin-reuptake inhibitor, and mianserin. Hyperthermia responded to parenteral sodium dantrolene and oral bromocriptine administration. The significant role of tetrabenazine (a central nervous system dopamine-depleting drug) and the contribution of antidepressants to the mechanism of this neuroleptic malignant syndrome - like hyperthermia are discussed.