In recent times, atypical antipsychotics are increasingly being used in the neonatal intensive care unit (NICU) for the management of neonatal delirium. As the recognition of delirium in NICU infants increases, caution should be exercised with use of antipsychotics for management, given associated adverse effects. Neuroleptic malignant syndrome (NMS) is a rare adverse drug reaction associated with exposure to antipsychotics and other antidopaminergic medications. Most reported cases of NMS in pediatric patients have been in older children on antipsychotic medications. We present a case of a 10-month-old former preterm infant who developed clinical signs suggestive of NMS after exposure to olanzapine for treatment of delirium. Our case report details the clinical course of this infant, delves into the condition, and outlines some useful lessons for the clinician in the identification and management of this rare but life-threatening adverse effect.

Key Points

NMS is a rare side effect of antipsychotic medications.

Hyperthermia with mental status changes could be due to NMS.

Antipsychotics should be used cautiously in infants.