(131) Atypical Presentation of Neuroleptic Malignant Syndrome in a Patient with Autism Spectrum Disorder Requiring ECT

Background: Many authors believe that catatonia and neuroleptic malignant syndrome (NMS) represent a single spectrum of illness (Fricchoine, 1985). Patients with autism spectrum disorder (ASD) may have catatonia-like features at baseline (Klek et al., 2023). Patients with ASD who are diagnosed with NMS may have physical exam findings that reflect both syndromes. We present a patient with ASD who presents with NMS that ultimately required ECT.  

Case: Mr. L is a 29-year-old male with a history of ASD, ADHD, and depression who presented with altered mental status and tachycardia. He had increasing agitation prior to admission, resulting in escalating doses of risperidone and haloperidol over three weeks. His baseline physical exam prior to hospitalization was unobtainable. He then developed significant fever, tachycardia, elevated CK, and lead-pipe rigidity, resulting in a diagnosis of NMS, but he had atypical findings of ankle clonus and hyperreflexia. He was initially given benzodiazepines, bromocriptine, and dantrolene as well as cyproheptadine. His condition then worsened, requiring intubation. ECT was quickly initiated for refractory NMS, even while workup was still ongoing. MRI, lumbar puncture, and EEG were benign. Tachycardia and fever improved, and rigidity gradually subsided. Initial presentation of ankle clonus and hyperreflexia did not resolve throughout the nine treatments of ECT and were thought to be part of his baseline exam. The patient was discharged one week after stopping ECT.

Discussion: ECT has long been indicated for cases of refractory NMS after failure of pharmacotherapy. Although significant differences in mortality have not been shown across all cases of NMS, treatment with ECT had 0% mortality, while bromocriptine and dantrolene had 8.5%, and supportive care alone showed 10.2% mortality (Kuhlwilm 2020). This highlights the necessity of urgent ECT for patients with refractory NMS, even at the expense of delaying diagnostic workup. The team had thought to obtain further diagnostic workup of refractory NMS given the presence of atypical physical exam findings and lack of improvement with pharmacological treatment. A baseline exam perhaps would have saved the expense/time of the testing. 

Conclusion:   ECT was effective for this patient with ASD who presented with NMS and an atypical physical exam. This case shows that medical teams should be mindful of exam findings that could be red herrings, and their workups should not delay the transition to ECT. Having a baseline exam could limit the need for extraneous workup.

References:

1. Kuhlwilm L, Schönfeldt-Lecuona C, Gahr M, Connemann BJ, Keller F, Sartorius A. The neuroleptic malignant syndrome-a systematic case series analysis focusing on therapy regimes and outcome. Acta Psychiatr Scand. 2020 Sep;142(3):233-241. doi: 10.1111/acps.13215. Epub 2020 Aug 2. PMID: 32659853.

2. Klek, Stefan., et al.  Neuroleptic Malignant Syndrome in a Patient with Autism Spectrum Disorder: Case Report. OBM Neurobiology. 2023; 07. 1-11. 10.21926/obm.neurobiol.2304188

3. Fricchione GL. Neuroleptic catatonia and its relationship to psychogenic catatonia. Biol Psychiatry. 1985 Mar;20(3):304-13. doi: 10.1016/0006-3223(85)90060-5. PMID: 2858225.

 

Presentation Eligibility: Not previously published or presented.

Diversity, Equity, and Inclusion: This abstract highlights a case of NMS in a patient who represents a population of patients with neurodevelopmental disorders. Patients with autism spectrum disorder frequently also have a co-morbid diagnosis of intellectual disabilities, which can be exclusionary to participation in clinical trials. Furthermore, there may be some communicative difficulties with patients with autism spectrum disorder in trials. For this reason, case reports, case series, and retrospective chart reviews provide valuable insight to clinicians about how to provide exceptional care for this patient population.