Abstract

Background: Children with congenital hemiplegia have difficulties reaching, grasping, manipulating, and releasing objects with their impaired upper limb (UL). There is now strong evidence supporting intensive models of UL therapy (e.g., constraint induced movement therapy [CIMT], bimanual training [BIM], goal-directed therapy) to improve UL function and achieve individualised goals. Despite increased knowledge regarding optimal protocols for these interventions, our understanding of individual and environmental factors influencing outcomes remains limited. It is known nevertheless that the more children embrace moderate challenges and persist with skill mastery the more likely they are to achieve functional goals. In the context of therapy, children’s willingness to persist with challenging tasks (mastery motivation) may be a previously unrecognised contributor to outcomes.Aims: The primary aim of this thesis was to determine the extent to which mastery motivation predicted occupational performance outcomes immediately post (at 13 weeks) and at 26 weeks following 2 different goal-directed UL interventions in school-aged children with congenital hemiplegia. Secondary aims were to: (1) systematically review clinimetric properties of measures of motivation; (2) examine test-retest reproducibility of parent proxy-report mastery motivation questionnaires; (3) examine relationships between mastery motivation, individual, and environmental characteristics and; (4) determine relationships between mastery motivation and engagement during therapy.Design and methodology: This study was embedded within a matched pairs randomised comparison trial COMBiT (Constraint induced Movement and Bimanual Therapy; NHMRC 1003887) for school-aged children with congenital hemiplegia. Fifty three children aged 5 to 16 years, Manual Ability Classification System levels I and II, were randomly allocated to receive either (1) Hybrid CIMT (hCIMT), an intensive group-based block model of modified CIMT followed by BIM (total dose 45 hours UL training) or (2) an individualised, distributed model of standard occupational therapy care (SC) (total dose 45 hours UL training). Mastery motivation was assessed at baseline using the Dimensions of Mastery Questionnaire (DMQ) parent proxy-report. Engagement in therapy was measured during goal-directed activities using the Pediatric Volitional Questionnaire (PVQ). Occupational performance outcomes were assessed at baseline, immediately following intervention (13 weeks), and at 26 weeks post-intervention using the Canadian Occupational Performance Measure (COPM). Repeated measurement of DMQ parent proxy-report over two interviews (2-30 days apart) enabled determination of test-retest reproducibility of the DMQ.Results: A systematic review identified 2 measures of motivation for school-aged children with physical disability or motor delay: the DMQ and the PVQ. Clinimetric review identified the DMQ to be the more robust of the 2 measures, however, further evidence of test-retest reproducibility and parent-child concordance of the DMQ was required. Test-retest reproducibility for DMQ instrumental aspect (ICC=0.86) and total motivation (ICC=0.84) were excellent with subscales ranging from ICC=0.70 to 0.91. Standard error of measurement (SEM) for total motivation was 0.23 points. Parent-child concordance was poor across all scores (ICC=-0.04 to 0.42) with large SEM (0.50-0.91). Associations between mastery motivation and individual and environmental factors, identified consistent and positive parental disciplinary practices were associated with higher total motivation (β=-0.19, p=0.01) and instrumental aspect scores (β=-0.20, p=0.01). Parental disciplinary laxness predicted decreased motivation. Children from single-parent families (β=0.88, p=0.004) and with one or more siblings (β=0.66, p=0.02) were more likely to react to failure. For occupational performance outcomes (COPM), both treatment groups made statistically and clinically significant changes in perceived performance and satisfaction of identified functional goals from baseline to 13 and 26 weeks post-intervention. No between-group clinically significant differences were identified. Superior bimanual performance (AHA: β=0.03, p<0.001), greater object-oriented persistence (DMQ: β=0.31, p=0.05), and being part of the SC treatment group (SC: β=0.24, p=0.01) predicted COPM performance scores at 13 and 26 weeks post-intervention. Despite parents reporting lower levels of mastery motivation for their children at baseline (mean diff=-0.25, p=0.05), children in hCIMT demonstrated greater engagement during goal-directed activities than children in SC (mean diff=0.24, p=0.04). For participants who received hCIMT, greater objectoriented persistence was associated with task-directedness (β=0.25, p=0.05), seeking challenges (β=0.51, p=0.02), exploration (β=0.10, p=0.03), and total volitional scores (β=0.23, p=0.01).Conclusions: Our understanding of contextual drivers of change in occupational performance within UL rehabilitation is limited. This thesis adds unique knowledge regarding the contribution of children’s individual characteristics and environmental factors to therapy outcomes. Children’s task persistence alongside bimanual performance, were identified as significant predictors of occupational performance outcomes following UL intervention for children with congenital hemiplegia. To support children’s task persistence in goal-directed activities, clinicians may consider designing therapy interventions that are reflective of children’s mastery motivation, individual strengths and family circumstances. Environments and strategies that are autonomysupportive, offer social support and scaffold the learning of new skills and achievement of personally meaningful goals can further maximise children's engagement and optimise their therapeutic outcomes. As part of early childhood interventions, clinicians may consider supporting parents to generalise these strategies within the home environment to foster children’s development of persistence when approaching difficult circumstances and activities.

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