A realist evaluation of a physical activity participation intervention for children and youth with disabilities: what works, for whom, in what circumstances, and how?

BACKGROUND: The need to identify strategies that facilitate involvement in physical activity for children and youth with disabilities is recognised as an urgent priority. This study aimed to describe the association between context, mechanisms and outcome(s) of a participation-focused physical activity intervention to understand what works, in what conditions, and how. METHODS: This study was designed as a realist evaluation. Participant recruitment occurred through purposive and theoretical sampling of children and parents participating in the Local Environment Model intervention at Beitostolen Healthsports Centre in Norway. Ethnographic methods comprising participant observation, interviews, and focus groups were employed over 15 weeks in the field. Data analysis was completed using the context-mechanism-outcome framework of realist evaluation. Context-mechanism-outcome connections were generated empirically from the data to create a model to indicate how the program activated mechanisms within the program context, to enable participation in physical activity. RESULTS: Thirty one children with a range of disabilities (mean age 12y 6 m (SD 2y 2 m); 18 males) and their parents (n = 44; 26 mothers and 18 fathers) participated in the study. Following data synthesis, a refined program theory comprising four context themes, five mechanisms, and six outcomes, were identified. The mechanisms (choice, fun, friends, specialised health professionals, and time) were activated in a context that was safe, social, learning-based and family-centred, to elicit outcomes across all levels of the International Classification of Functioning, Disability and Health. CONCLUSIONS: The interaction of mechanisms and context as a whole facilitated meaningful outcomes for children and youth with disabilities, and their parents. Whilst optimising participation in physical activity is a primary outcome of the Local Environment Model, the refined program theory suggests the participation-focused approach may act as a catalyst to promote a range of outcomes. Findings from this study may inform future interventions attempting to enable participation in physical activity for children and youth with disabilities.

Associations between recurrent musculoskeletal pain and visits to the family doctor (GP) and specialist multi-professional team in 74 Norwegian youth with cerebral palsy.

BACKGROUND: Musculoskeletal pain in cerebral palsy (CP) is common, but probably undertreated. The aim of the study was to explore if recurrent musculoskeletal pain (RMP) related to the CP condition was associated with visits to the family doctor [general practitioner (GP)] and specialist habilitation centre in youth with CP. METHODS: Seventy-four youth with CP (mean age 16.5 years, 40 boys) from the same geographical area were assessed by clinical examination, semi-structured interview on pain (adolescent and parent together), the two questions on pain from the Child Health Questionnaire (parents only) and a structured interview on health care services. Gross Motor Function Classification System was level I 39%, level II 23%, level III 8% and levels IV and V 30%. RESULTS: Thirty-five participants (47%) had visited their GP last year, and 49 (66%) had visited the specialist habilitation centre. The presence of RMP (n = 58; 78%) was not significantly associated with having visited the GP or the specialist habilitation centre. Still, in participants with RMP, increasing pain severity was associated with having visited the GP. Rest, massage, change of position and oral drugs were the most common measures taken to relieve RMP. Three adolescents with RMP did not take any measures to relieve pain. CONCLUSION: Youth with RMP do take measures to relieve pain, but usually not in the direction of consulting the health care services available unless the pain is severe. Both youth with CP and their caregivers should be encouraged to discuss RMP with their professional network of care.

Walking deterioration and gait analysis in adults with spastic bilateral cerebral palsy.

Walking deterioration occurs frequently in adults with spastic bilateral cerebral palsy (CP), but their gait characteristics are largely unknown. The study aims were (1) to compare selected gait analysis variables between those reporting and those not reporting walking deterioration, and (2) to characterise the overall gait deviations and classify the gait patterns. Participants (N=16) were recruited from a follow-up study, had spastic bilateral CP, <40 years in 2006, GMFCS levels I-III, and could walk at least 10 m without support. Eight reported walking deterioration (cases) and eight did not (controls). A theoretical framework linking work of walking, fatigue and deterioration in walking was developed. It was hypothesised that higher energy requirements during gait and larger gait deviations would be associated with deterioration in walking. Three-dimensional gait analysis was used to obtain centre of mass work, mechanical joint work, lower limb kinematics, movement analysis profile (MAP), and gait profile scores (GPS). There were no differences between the cases and controls in centre of mass work, joint work, or in the GPS. The largest MAP deviations were seen in sagittal pelvis, hip, and knee angles and foot progression. Crouch and asymmetric gait were common patterns. Walking deterioration could not be explained by these work and kinematic variables. An individual's perception of deterioration in walking is subjective, and may be experienced and interpreted differently across people. Larger, longitudinal studies on the natural history of walking in spastic CP are needed. Qualitative studies on the subjective experiences of walking deterioration are also warranted.

An international comparison of patterns of participation in leisure activities for children with and without disabilities in Sweden, Norway and the Netherlands.

PURPOSE: To investigate whether there are differences in participation in leisure activities between children with and without disabilities in Sweden, Norway and the Netherlands and how much personal and environmental factors explain leisure performance. METHODS: In a cross-sectional analytic design, the Children's Assessment of Participation and Enjoyment, CAPE, was performed with 278 children with disabilities and 599 children without disabilities aged 6-17 years. A one-way between-groups ANOVA explored the differences in participation between the countries. Hierarchical multiple regression analysis assessed if age, gender, educational level, living area and country of residence explained the variance in participation. RESULTS: Scandinavian children with disabilities participated in more activities with higher frequency compared to Dutch children. The strongest predictor was country of residence. For children without disabilities, differences existed in informal activities, the strongest predictor was gender. CONCLUSION: Differences in school- and support systems between the countries seem to influence patterns of participation, affecting children with disabilities most.

Continuous intrathecal baclofen therapy in children with cerebral palsy - when does improvement emerge?

AIM: The aim of the study was to explore the timing of effects of intrathecal baclofen therapy in children with cerebral palsy. METHODS: Thirty five children with severe disabilities with cerebral palsy who started continuous intrathecal baclofen therapy (CITB) were followed for 18 months. Pain, number of awakenings during night, spasticity, GMFM-66 scores and PEDI scores were recorded the day before pump implantation and after 6 and 18 months of treatment respectively. RESULTS: Introduction of CITB was associated with changes across all ICF dimensions. Reduced pain and improved sleep occurred within 6 months of treatment. Social function improved within 6 months and continued to improve until 18 months of CITB. Mobility also improved, but with a latency. CONCLUSION: There seems to be a sequence of changes after introduction of continuous intrathecal baclofen in a child with cerebral palsy that may guide the multidisciplinary team in their timing of therapy during post-surgical follow-up.

Locomotion skills in adults with cerebral palsy.

OBJECTIVES: To investigate self-reported locomotion skills in persons with cerebral palsy (CP) and to investigate variables potentially associated with deterioration of walking skills. DESIGN: Cross-sectional retrospective survey. SETTING AND SUBJECTS: A multidimensional mailed questionnaire was sent to 766 persons with CP, 18 years or over, without intellectual disabilities, living in Norway. MAIN OUTCOME MEASURES: The questionnaire consisted of demographic and diagnostic items, items on locomotion skills, and physical function (SF-36). RESULTS: In total 406 persons, 51% males and 49% females from 18 to 72 years (mean 34 years, SD 11 years) with all categories of CP responded. Median age for reported walking debut was 3 years, with a range from 1 to 14 years. In total 216 respondents (53%) walked without support, 104 persons (25%) walked with support, 39 persons (10%) had lost their walking skills, and 47 (12%) had never been able to walk. Mean level of physical function (SF-36) was 53 out of 100. There were 97 persons (27%) who reported improvement of walking skills, mainly before 25 years, 102 (28%) reported no change, and 160 (44%) reported deterioration, mainly before 35 years of age. Deterioration was significantly associated with older age, delayed walking debut and severe neurological impairment. Self-reported causes of deterioration were pain, fatigue and lack of adapted physical activity. CONCLUSION: Deterioration of locomotion skills is a significant problem in persons with CP from an early age, documenting the need for life-long follow-up. The predictors above should be investigated in further clinical studies, searching for potential causal pathways.

Coping potential and disability--sense of coherence in adults with cerebral palsy.

PURPOSE: The aim of the present study was to investigate sense of coherence (SOC) in adults with cerebral palsy (CP) compared to the general population. METHOD: A questionnaire was sent to a representative sample of adults with CP, including the 3-item version of SOC and items on their life situation and follow-up programmes. The study included persons over 18 years of age with CP and without intellectual disability. The results were compared with the results from a reference group. RESULTS: The 406 respondents with CP (48.5% females, 51.5% males) were 18-72 years of age. The distribution of the different types of CP coincided with international epidemiological studies. Mean SOC was significantly lower in the adults with CP than in the reference group. The largest difference was found in the domain of comprehensibility. The factors most important in relation to SOC were level of education, marital status, life satisfaction and fatigue. CONCLUSIONS: Early experiences of predictability, balance between challenges and personal resources and finding these challenges worthy of investment, are prerequisites for developing sense of coherence. The present study suggests that these factors are less present in early socialization of persons with CP compared to the general population, and underlines the need for follow-up programmes that emphasize existential aspects and coping strategies.

Effects of protein-degradation inhibitors on the inactivation of tyrosine aminotransferase, tryptophan oxygenase and benzopyrene hydroxylase in isolated rat hepatocytes.

The following three potent inhibitors of hepatocytic proteolysis were investigated to see if they would inhibit the intracellular inactivation of enzymes: chymostatin and leupeptin (proteinase inhibitors) and methylamine (a lysosomotropic weak base). Chymostatin inhibited the inactivation of two of the three enzymes tested: tyrosine aminotransferase (EC 2.6.1.5) and tryptophan oxygenase (tryptophan 2,3-dioxygenase, EC 1.13.11.11). Leupeptin had no effect on any of the enzymes, whereas methylamine had only a weak inhibitory effect on tyrosine aminotransferase inactivation. Apparently proteolytic cleavage (probably by a non-lysosomal proteinase, since only chymostatin is effective) is involved in the inactivation of tyrosine aminotransferase and tryptophan oxygenase. The third enzyme, benzopyrene hydroxylase (flavoprotein-linked mono-oxygenase, EC 1.14.14.1), is probably inactivated by a non-proteolytic mechanism.