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Zuclopenthixol is usually used in parental form to manage acute agitation and psychosis.[ 1 ] It has high affinity for dopamine D1 and D2 receptors. There are very few reports of Neuroleptic Malignant Syndrome (NMS) with use of zuclopenthixol monotherapy. In this case report, we present a 35 year old male with alcohol dependence, presented to the emergency with altered sensorium, fever and stiffness of limbs. He had history of receiving Injection Zuclopenthixol acetate 200 mg thrice over 24 hours. Within 12-14 hours of the last injection, patient developed features suggestive of NMS. On investigations he was found to have raised serum creatinine phosphokinase levels (839.9U/L; reference laboratory value: 26 to 308 U/L) and leukocytosis. In view of these features, he was diagnosed with NMS and was started on supportive management to address the dehydration and was given Thiamine 500 mg thrice daily. Additionally he was started on Tab. Bromocriptine 5 mg thrice daily and lorazepam 2 mg/day. With this intervention, his symptoms improved over the period of 1 week and tablet bromocriptine was tapered off, after 1 week of being asymptomatic.

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