Neuroleptic Malignant Syndrome in Children with Autism Spectrum Disorder (ASD): A Case Report and Brief Review of Recent Literature

Stefano Berloffa,Ilaria Lupetti,Benedetta Tascini,Beatrice Fossati,Claudia Dosi,Gabriele Masi

doi:10.3390/children8121201

Abstract

Neuroleptic malignant syndrome (NMS) is a rare, life-threatening, idiosyncratic adverse reaction to antipsychotic drugs. Despite the increasing rates in the prescription of antipsychotics in pediatric patients with autism spectrum disorder (ASD), little is known about the occurrence and hallmarks of NMS in this specific population. NMS appears to be part of the larger catatonia domain, based on the frequent relationship between ASD and catatonia, on the shared, when not overlapping, clinical features with malignant catatonia, and on the effectiveness of catatonia treatments on the NMS/MC symptoms. The intrinsic difficulties of exploring NMS in ASD in controlled studies accounts for the subsequent lack of available information. Based on recent reports and on our case report, clinical features of NMS in the pediatric ASD population appear to be the same as the non-ASD population. Further studies are needed to confirm these results.

Highlights

Neuroleptic malignant syndrome (NMS) is a rare, life-threatening, iatrogenic condition mainly associated with antipsychotic drug use, and is characterized by fever, altered mental status, muscle rigidity, and autonomic dysfunction
A non-alternative model proposes the concomitant role of toxicity of the musculoskeletal fibers, supported by the response to dantrolene in NMS and typical antipsychotic drug effects on calcium regulation in skeletal muscular fibers [2]
We describe the case of an 18-year-old boy presented with autism spectrum disorder (ASD) associated with a mild intellectual disability

Summary

Introduction

Neuroleptic malignant syndrome (NMS) is a rare, life-threatening, iatrogenic condition mainly associated with antipsychotic drug use, and is characterized by fever, altered mental status, muscle rigidity, and autonomic dysfunction. DSM-5 describes the most common clinical features, which include a positive history for dopamine antagonist use (usually within 72 h prior to symptom development), as hyperthermia, generalized rigidity, described as “lead pipe” in its most severe form, and other neurological and medical symptoms (e.g., tremor, sialorrhea, akinesia, dystonia, trismus, myoclonus, dysarthria, dysphagia, and rhabdomyolysis) They include, in most cases, changes in mental status, characterized by delirium or altered consciousness (often an early sign), and autonomic instability (tachycardia, diaphoresis, blood pressure elevation or fluctuation, urinary incontinence, and tachypnea). A massive asymptomatic CK elevation has been described during antipsychotic exposure in nonpsychotic, drug-naïve youth during treatment with second-generation antipsychotics In these cases, a drug discontinuation should be considered only when possible signs of NMS or rhabdomyolysis are suspected (i.e., flulike syndrome, fever, weakness, alteration of consciousness, muscle rigidity, tachycardia, hyper-/hypotension, and dark urine) [11]. Data on other treatments for catatonia in children with ASD are still lacking, except for a recent case report including two patients successfully treated with clozapine [20]

Case Presentation

Discussion