King's College London

Research portal

Rehabilitation in childhood-onset hyperkinetic movement disorders including dystonia: Treatment change in outcomes across the ICF and feasibility of outcomes for full trial evaluation

Research output: Contribution to journalArticlepeer-review

Hortensia Gimeno, Helene J. Polatajko, Victoria Cornelius, Jean Pierre Lin, Richard G. Brown

Original languageEnglish
Pages (from-to)159-167
Number of pages9
JournalEuropean Journal of Paediatric Neurology
Volume33
DOIs
Accepted/In press2021
PublishedJul 2021

Bibliographical note

Funding Information: We would like to thank the children, young people, and their families that participated in the study. We would also like to thank the blind raters, Professor Gill Chard from AMPS UK and Ireland; Allied Health Professionals from the Complex Motor Disorders Service, Kylee Tustin, Erin Morton, Lesley Baker, Laura Ainaga and Emily Fleischfresser. Hortensia Gimeno is funded by a National Institute for Health Research (NIHR/HEE Clinical Doctoral Research Fellowship, CDRF-2013-04-039 ). This paper represents independent research part funded by the National Institute for Health Research (NIHR) (HG), and the NIHR Biomedical Research Centre at South London and Maudsley NHS Foundation Trust and King's College London (RB). The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. Funding Information: We would like to thank the children, young people, and their families that participated in the study. We would also like to thank the blind raters, Professor Gill Chard from AMPS UK and Ireland; Allied Health Professionals from the Complex Motor Disorders Service, Kylee Tustin, Erin Morton, Lesley Baker, Laura Ainaga and Emily Fleischfresser. Hortensia Gimeno is funded by a National Institute for Health Research (NIHR/HEE Clinical Doctoral Research Fellowship, CDRF-2013-04-039). This paper represents independent research part funded by the National Institute for Health Research (NIHR) (HG), and the NIHR Biomedical Research Centre at South London and Maudsley NHS Foundation Trust and King's College London (RB). The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. Publisher Copyright: © 2021 Copyright: Copyright 2021 Elsevier B.V., All rights reserved.

King's Authors

Abstract

Background: Childhood-onset hyperkinetic movement disorders (HMD), including dystonia are notoriously difficult to treat and there are limited studies showing successful medical, surgical or non-pharmacological interventions. Methods: This prospective study used grouped data (n = 22) from two studies of the Cognitive Orientation to daily Occupational Performance (CO-OP) Approach for patient-selected goals. Eligibility included aged 6–21 years, deep brain stimulation in place, with manual ability classification system level I-IV. Outcome was assessed on a range of patient-reported and clinician-rated measures across the International Classification of Function at end-treatment (10 weekly sessions) (series 1 and 2) and 3-month follow-up (series 1). Feasibility of outcomes to be used in a full trial were explored. Findings: Nineteen participants completed the intervention and were included in the analysis. Of the primary outcome measures, the self-reported Canadian Occupational Performance Measure showed improvement in goal performance (mean change 4.08, 95% CI [3.37,4.79] post-; 4.18 [5.10,5.26] follow-up), and satisfaction (4.03 [3.04,5.03) post-; 4.44 [3.07,5.82] follow-up]. The Assessment of Motor and Process Skills showed improved motor score (0.52 [0.01,1.03] at follow-up only, while the process score did not change. Objective blind-rated pooled data using the Performance Quality Rating Scale-individualized indicated significant change for trained goals (3.79 [3.37,4.21] post-; (4.01,5.10) follow-up] and untrained goals (1.90 [1.24,2.55] post 1.91 [0.23,3.60] follow-up]. Motor impairment assessed by the Burke-Fahn Motor Disability Rating Scale was unchanged (−3.26 [-6.62,0.09] post-; −1.11 [-8.05,5.82] follow-up). Improvement was also observed in self-efficacy (0.97 [0.47,1.47] post-; 1.37 [1.91–0.83] follow-up) and Quality of Life (0.12 [0.03–0.22] follow-up). Goal improvement; self-efficacy and quality of life captured significant change post-intervention. This improvement was shown despite no change on impairment-related measures and were shown to be feasible measures to use in a larger study of CO-OP for this population.

View graph of relations

© 2020 King's College London | Strand | London WC2R 2LS | England | United Kingdom | Tel +44 (0)20 7836 5454