Abstract

Objective: To explore preliminary effectiveness of the Cognitive Orientation to daily Occupational Performance (CO-OP) Approach in improving outcomes in childhood-onset hyperkinetic movement disorders (HMDs) including dyskinetic cerebral palsy following deep brain stimulation (DBS) across UK clinical occupational therapists.Methods: Randomized, multiple-baseline, Single Case Experimental Design N-of-1 trial with replications across participants. Five self-selected goals were identified: three goals were worked on during CO-OP and two goals were left untreated and used to assess skills transfer. Participants were between 6 and 21 years and had received DBS surgery with baseline Manual Ability Classification System (MACS) levels I–IV. Participants were randomized to typical or extended baseline (2 vs. 6 weeks), followed by 10 weekly individual CO-OP sessions. The primary outcome was functional performance measured by the Performance Quality Rating Scale-Individualized (PQRS-I), assessed before, during, and following treatment. Outcome assessors were blinded to baseline allocation, session number, and assessment time. A non-overlapping index, Tau-U, was used to measure effect size.Results: Of the 12 participants recruited, 10 commenced and completed treatment. In total, 63% of trained goals improved with effect sizes 0.66–1.00 (“moderate” to “large” effect), seen for all children in at least one goal. Skills transfer was found in 37% of the untrained goals in six participants.Conclusions: Cognitive strategy use improved participant-selected functional goals in childhood-onset HMD, more than just practice during baseline. Preliminary effectiveness is shown when the intervention is delivered in clinical practice by different therapists in routine clinical settings.

Highlights

  • METHODSIn children and young people with a hyperkinetic movement disorder (HMD) including dyskinetic cerebral palsy (CP) surgical treatments such as deep brain stimulation (DBS) can be effective in reducing severity of motor impairments [1, 2]

  • We have recently reported a proof-of-concept efficacy study [7] of the Cognitive Orientation to daily Occupational Performance (CO-OP) [8] used with children and young people with HMD and DBS in place

  • Duration since DBS ranged from 6 months to 4 years 6 months

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Summary

Introduction

METHODSIn children and young people with a hyperkinetic movement disorder (HMD) including dyskinetic cerebral palsy (CP) surgical treatments such as deep brain stimulation (DBS) can be effective in reducing severity of motor impairments (e.g., dystonia) [1, 2]. A recent systematic review of interventions for CP indicated strong evidence that surgical interventions, such as intrathecal baclofen infusion pump or selective dorsal rhizotomy, and medical interventions, such as botulinum toxin injections, reduce tone in children with spastic CP [4]. It is the combination of these interventions with adjunct rehabilitation such as strength training or occupational therapy that yield effective results in improving motor and/or functional outcomes. Number of cases are relatively small, and the heterogeneity of these disorders in terms of etiology, motor severity, and non-motor factors make the planning and implementation of large scale randomized controlled trials (RCTs) challenging

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