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Dystonia in children with acquired brain injury

Published:September 26, 2022DOI:https://doi.org/10.1016/j.ejpn.2022.09.004

      Highlights

      • The estimated incidence of dystonia following ABI admitted to the PICU was 3.2%.
      • The incidence was higher in the hypoxia/anoxia (8.3%) and TBI (6.2%) cohorts.
      • Dystonia emerged early (all within one month) and persisted at 6 months in the majority.
      • Patients who developed dystonia had median lower GCS and median longer PICU admission.

      Abstract

      Aim

      To quantify the proportion of children who develop dystonia after acquired brain injury (ABI) admitted to a tertiary paediatric intensive care unit (PICU) and analyse the trajectory of dystonia over a 6 month period.

      Methods

      Children's Health Ireland at Temple Street PICU electronic database was searched for key terms related to ABI from January 1, 2016 to March 14, 2021. Individuals meeting inclusion criteria were analysed, and clinical data pertinent to ABI, dystonia, treatment and outcomes were reviewed.

      Results

      Six-hundred and forty-three PICU episodes (580 patients) met search criteria for ABI, with 379 included in the final analysis. Twelve patients developed dystonia following ABI, giving an incidence of 3.2%. The incidence was higher in the hypoxia/anoxia and TBI cohort at 8.3% and 6.2%, respectively. All patients developed dystonia within the first month following ABI (50% by a week). Patients who developed dystonia compared to non-dystonia cohort had a median lower GCS on admission (4.5 versus 7.0, p value 0.032), longer median length of PICU stay (14.0 versus 3.0 days, p value < 0.001) and were older (median age 9.08 versus 4.68 years, p value 0.06). Dystonia persisted in the majority at 6 months (10/11), requiring on-going medical therapies.

      Conclusion

      In our retrospective study, the estimated incidence of dystonia following ABI admitted to the PICU was 3.2%, highest in the hypoxia/anoxia (8.3%) and TBI (6.2%) cohorts. Dystonia emerged early and persisted at 6 months in the majority. This is the first review of dystonia, clinical trajectory and outcomes conducted post-PICU admission for ABI. Future prospective studies are required to determine the true prevalence and burden of disease in the PICU setting.

      Abbreviations:

      ABI (Acquired brain injury), CK (Creatinine kinase), CNS (Central nervous system), GCS (Glasgow coma scale), IQR (Inter-quartile range), PICU (Paediatric intensive care unit), PSH (Paroxysmal Sympathetic Hyperactivity), TBI (Traumatic brain injury)
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