PT - JOURNAL ARTICLE AU - Gimeno, Hortensia AU - Brown, Richard G. AU - Lin, Jean-Pierre AU - Cornelius, Victoria AU - Polatajko, Helene J. TI - Cognitive approach to rehabilitation in children with hyperkinetic movement disorders post-DBS AID - 10.1212/WNL.0000000000007092 DP - 2019 Mar 12 TA - Neurology PG - e1212--e1224 VI - 92 IP - 11 4099 - http://n.neurology.org/content/92/11/e1212.short 4100 - http://n.neurology.org/content/92/11/e1212.full SO - Neurology2019 Mar 12; 92 AB - Objective This proof-of-concept feasibility trial examined the potential of the Cognitive Orientation to daily Occupational Performance Approach (CO-OP) to augment deep brain stimulation (DBS) outcomes in childhood-onset hyperkinetic movement disorders (HMD) including dystonia and dyskinetic cerebral palsy.Methods This is a single case experimental design using multiple baseline as n-of-1 trial comprising 10 intervention sessions, with replications across participants (n = 10). Treatment focused on 3 participant-selected goals. Transfer was assessed on 2 additional untreated goals. Individuals enrolled were 6–21 years of age and had DBS in situ and sufficient manual ability. Primary outcome was functional performance change on the Performance Quality Rating Scale–Individualized (PQRS-i) measured before, during, and posttreatment, and at 3-month follow-up. Assessors of outcome were blinded to time of assessment, number of intervention session, and treatment allocation. To measure effect size, a nonoverlapping index, Tau-U, was used. Feasibility measures were captured.Results One participant withdrew before baseline assessment. Effect sizes of at least 0.66 were seen at both posttreatment and follow-up with all participants showing improvements in at least one trained goal in PQRS-i. Six participants improved on all 3 goals and 2 improved on 2 trained goals. Two children showed deterioration in one trained goal each. Transfer to untrained goals was observed in 3 participants for a total of 5 goals. CO-OP was feasible and acceptable to all participants.Conclusion A cognitive-based, task-oriented approach to support performance of personally relevant functional skills enabling participation is acceptable in childhood-onset HMD post-DBS. Further, preliminary efficacy to improve outcomes and proof of concept with CO-OP has been established in this population.Classification of evidence This study provides Class IV evidence that for children with HMD who had undergone DBS, CO-OP improves performance of personally relevant functional skills.CO-OP=Cognitive Orientation to daily Occupational Performance Approach; COPM=Canadian Occupational Performance Measure; CP=cerebral palsy; DBS=deep brain stimulation; HMD=hyperkinetic movement disorders; OLS=ordinary least squares; PQRS-I=Performance Quality Rating Scale–individualized; SCED=single-case experimental design; SCRIBE=Single Case Reporting Guideline in Behavioural Interventions