Catatonia and neuroleptic malignant syndrome in patients with cerebral palsy: Two case reports and a systematic review of the literature.

BACKGROUND: Patients with cerebral palsy, a group of movement disorders with motor, communication, and behavioral features that can mimic catatonic signs, could benefit from efforts to improve detection and treatment of comorbid catatonia. Given that cerebral palsy frequently co-occurs with conditions associated with catatonia such as autism spectrum disorder, epilepsy, intellectual disability, and mood and psychotic disorders, lifetime prevalence of catatonia in this population may be high.

OBJECTIVE: To systematically review the literature on catatonia and the related condition of neuroleptic malignant syndrome (NMS) in patients with cerebral palsy, while presenting two additional cases of catatonia.

METHODS: We used the terms "cerebral palsy" in combination with "catatoni*", related terms for catatonia, and "neuroleptic malignant syndrome" to query OVID Medline (1948 - November 28, 2022), PsycINFO, Cumulative Index to Nursing, and Allied Health Literature, and Embase for applicable case reports. The Neuroleptic Malignant Syndrome Information Service database was also manually searched.

RESULTS: In addition to our two catatonia reports, we identified ten reports of catatonia in patients with cerebral palsy, as well as eight reports of NMS. Patients with both conditions responded well and, sometimes rapidly, to treatment. Notably, of the five patients with catatonia and cerebral palsy who received electroconvulsive therapy (ECT), two developed recurrent self-limited hyperthermia post-treatment. We also identified several cases of baclofen withdrawal, which can be life threatening due to seizure risk, presenting with NMS-like features in patients with cerebral palsy who had malfunctioning intrathecal baclofen pumps for spasticity management.

CONCLUSIONS: Given frequent comorbidity of conditions associated with catatonia in patients with cerebral palsy, as well as routine treatment with medications that can induce NMS, such as metoclopramide and anticholinergics, catatonia and NMS may be underreported in the cerebral palsy patient population, despite being highly treatable. Possible underdiagnosis of catatonia in patients with cerebral palsy may be due to misattribution of overlapping features between the two conditions to cerebral palsy. Clinicians should be aware of possible recurrent self-limited fever when employing ECT to treat patients with catatonia and cerebral palsy, while also being vigilant for intrathecal baclofen withdrawal when encountering NMS-like features in patients with cerebral palsy.