Fatigue experienced by people with cerebral palsy: a systematic review of assessment tools and decision tree.

PURPOSE: To conduct a systematic review of self- and proxy-report fatigue assessment tools used in studies of people with cerebral palsy (CP) of all ages, and to develop a fatigue assessment tool decision tree for clinicians and researchers. MATERIALS AND METHODS: Five electronic databases (MEDLINE, PsycInfo, CINAHL, Web of Science and Cochrane) were searched to September 2021 to identify studies assessing self-reported fatigue in people with CP of any age. The assessment tools utilised were extracted and two reviewers appraised the tool characteristics, clinical utility and psychometric properties. A decision tree for selecting fatigue assessment tools was constructed. RESULTS: Ten assessment tools were identified across thirty-nine studies, three of which are valid and reliable for assessing fatigue severity and impact in people with CP. A four-level fatigue assessment tool decision tree was constructed. No valid and reliable tool for assessing cognitive fatigue was identified; responsiveness has not been evaluated in any tool for people with CP. CONCLUSIONS: Physical fatigue screening and assessment tools for people with CP are available and are presented in our decision tree, however their utility as outcome measures remains unclear. Cognitive fatigue is understudied and poorly understood, further work is required in this area.

Current measurement tools to screen and assess physical fatigue in people with cerebral palsy (CP) are valid and reliable and are presented in our 4-level decision tree to guide assessment tool selection.The responsiveness of these measurement tools to screen and assess physical fatigue has not been evaluated, therefore their utility as outcome measures in people with CP is unclear.Cognitive fatigue is understudied and poorly understood in people with CP.Valid and reliable tools to assess cognitive fatigue in people with CP are not available.

Maternal and child factors associated with the health-promoting behaviours of mothers of children with a developmental disability.

BACKGROUND/AIM: Mothers caring for their child or adult with a developmental disability can experience mental health disparity. Protective factors such as healthy behaviours are under-researched. This study investigated relationships between mental health, healthy behaviours, and disability factors. METHODS: The cross-sectional online survey included: Depression Anxiety Stress Scales (DASS); Family Empowerment Scale (FES); Health Promoting Activities Scale (HPAS); and a measure of childhood quality of life (QoL). RESULTS: All mothers were raising offspring (aged 3-36 years) with a developmental disability. Fifty-two percent of mothers (N = 81) had a mental health diagnosis. DASS scores were elevated for depression (58 %), anxiety (52 %) and stress (68 %). Mothers participated in health promoting activities infrequently and reported low satisfaction with community health-supporting facilities. Depressive symptoms, maternal empowerment and two indicators of child-related QoL explained 29.7 % of the variance in healthy behaviours. Depressive symptoms were the most important predictor of lack of health promoting behaviours. CONCLUSIONS: Better mental health predicted more frequent participation in health promoting behaviour. Future research might explore the extent to which health promoting behaviours protect mental health. Service changes including family health focused services, and custom designed health promotion or coaching programs may improve the health behaviours of mothers with high care responsibilities.

Use of health services by preschool-aged children who are developmentally vulnerable and socioeconomically disadvantaged: testing the inverse care law.

AIM: The inverse care law suggests that those with the greatest need for services are least likely to receive them. Our aim of this study was to test the inverse care law in relation to the use of health services by children aged 4-5 years in Australia who were developmentally vulnerable and socioeconomically disadvantaged. METHOD: Cross-sectional data were collected from the Longitudinal Study of Australian Children birth cohort when the children were aged 4-5 years. Children were grouped according to the combination of developmental vulnerability (yes, no) and socioeconomic disadvantage (lower, higher), resulting in four groups (reference group: developmentally vulnerable and disadvantaged). Multivariate regression was used to examine the impact of the combination of developmental vulnerability and disadvantage on health service use, adjusting for other sociodemographic characteristics. RESULTS: 3967 (90%) of children had data on developmental vulnerability at 4-5 years. A third of children (32.6%) were classified as developmentally vulnerable, and 10%-25% of these children had used health services. Non-disadvantaged children who were developmentally vulnerable (middle need) had 1.4-2.0 times greater odds of using primary healthcare, specialist and hospital services; and non-disadvantaged children who were not developmentally vulnerable (lowest need) had 1.6-1.8 times greater odds of using primary healthcare services, compared with children who were developmentally vulnerable and disadvantaged (highest need). CONCLUSION: We found some evidence of the inverse care law. Equity in service delivery remains a challenge that is critically important to tackle in ensuring a healthy start for children.

Health-related quality of life and upper-limb impairment in children with cerebral palsy: developing a mapping algorithm.

AIM: To: (1) investigate the relationship between upper-limb impairment and health-related quality of life (HRQoL) for children with cerebral palsy and (2) develop a mapping algorithm from the Cerebral Palsy Quality of Life Questionnaire for Children (CPQoL-Child) onto the Child Health Utility 9D (CHU9D) measure. METHOD: The associations between physical and upper-limb classifications and HRQoL of 76 children (40 females, 36 males) aged 6 to 15 years (mean age 9 years 7 months [SD 3y]) were assessed. Five statistical techniques were developed and tested, which predicted the CHU9D scores from the CPQoL-Child total/domain scores, age, and sex. RESULTS: Most participants had mild impairments. The Manual Ability Classification System (MACS) level was significantly negatively correlated with CHU9D and CPQoL-Child (r=-0.388 and r=-0.464 respectively). There was a negative correlation between the Neurological Hand Deformity Classification (NHDC) and CPQoL-Child (r=-0.476, p<0.05). The generalized linear model with participation, pain domain, and age had the highest predictive accuracy. INTERPRETATION: The weak negative correlations between classification levels and HRQoL measures may be explained by the restricted range of impairment levels of the participants. The MACS and NHDC explained the impact of upper-limb impairment on HRQoL better than the other classifications. The generalized linear model with participation, pain, and age is the suggested mapping algorithm. The suggested mapping algorithm will facilitate the use of CPQoL-Child for economic evaluation and can be used to conduct cost-utility analyses. WHAT THIS PAPER ADDS: The Manual Ability Classification System and Neurological Hand Deformity Classification were the best predictors of health-related quality of life measures. Age and Cerebral Palsy Quality of Life Questionnaire for Children participation and pain domain scores can predict Child Health Utility 9D scores.

A Rights-Based Approach for Service Providers to Measure the Quality of Life of Children with a Disability.

BACKGROUND: This paper identifies the best instruments for service providers to measure the quality of life (QoL) of children with a disability, with a focus on their alignment with the Convention on the Rights of Persons with a Disability (CRPD). METHODS: This study reviewed systematic reviews to identify generic QoL instruments for children and adolescents, followed by an appraisal process using newly developed criteria. QoL instruments with a health status, functioning, and condition-specific focus were excluded. RESULTS: Twenty generic QoL instruments for children were identified from existing systematic reviews to undergo further review. Only 2 of the 20 instruments were recommended for service providers to measure the QoL of children with a disability (KIDSCREEN and KINDL). Many pediatric QoL instruments (N = 9) focus on functioning and are not consistent with the CRPD, confounding a child's functioning with their feelings about their life. KIDSCREEN and KINDL have self-report and parent report versions, are applicable for childhood and adolescence, demonstrate adequate reliability and validity, involved children in their development, focus on wellbeing, are likely to be able to be completed by a child with a disability, and are low in cost. CONCLUSIONS: Many instruments focus on functioning rather than wellbeing and thus may not capture the QoL of children with a disability. A child's functional limitations may not be consistent with their feelings about life. Two instruments that assess wellbeing and meet the criteria important for service providers now require further testing to explore their usefulness and validity for children with varying abilities.

Supporting the mental health of mothers of children with a disability: Health professional perceptions of need, role, and challenges.

BACKGROUND: Mothers of children with a disability have a higher risk of mental health difficulties than mothers of typically developing children. Very little is known about how health professionals perceive their role in supporting mothers' mental health. We aimed to explore the perspectives of health professionals working with families of children with a disability about how they provide support for maternal mental health in their roles. Specifically, whether professionals consider it their role and responsibility to provide support, the types of actions that they engage in to do this, and the challenges that they experience. METHODS: This qualitative semi-structured interview study included 13 health professionals (allied health professionals, general practitioners, and paediatricians) working with families of a child with a disability. Thematic analysis was conducted on transcribed interview data. RESULTS: Four overlapping themes were identified from the data indicating that professionals knew that mothers needed mental health support but were not always clear about their roles and responsibilities to support maternal mental health. Professionals also found it difficult to address maternal mental health difficulties, were not always aware of the best strategies to support maternal mental health, and faced difficulties that could be overcome with training and system improvements. CONCLUSIONS: Although all health professionals were aware of the frequent occurrence of maternal mental health difficulties and the importance of addressing them, several challenges were identified to managing them successfully. Providing health professionals with training in discussing mental health and clearer referral pathways would contribute to mothers being better supported, in addition to policy change that allows parental support in child health services.

Systematic review of the economic impact of cerebral palsy.

BACKGROUND: Cerebral palsy (CP) and its associated conditions can pose a significant economic burden on families, the health care system and the general economy. The boundary for inclusion of costs in research can vary substantially across studies. AIMS: To summarize the evidence for burden of disease for CP including the impacts on the health system, the community and carers. METHODS: Literature was identified from Ovid Medline, Embase, CINHAL, PsyInfo, Econlit, Health Economic Evaluation Database (HEED) and NHS Economic Evaluation Database (NHS EED) in the Cochrane Library. The search was restricted to articles published in English between 1970 and April 2016. All costs were converted to $USD 2016 price. RESULTS: Twenty-two articles were included. Studies varied from snapshot cost descriptions to more complex lifetime estimates, from prevalence-based to incidence-based studies, and from inclusion to exclusion of non-medical costs. There was a strong positive relationship between CP severity and expenditure. Significant costs were incurred by families and the welfare system to facilitate school and community engagement. CONCLUSION: Facilitating participation for people with CP involves substantial expense. The size, nature and distribution of the economic burden emphasises the importance of finding effective strategies to reduce the risk and severity of CP, together with how it is financed.

Economic evaluation and cost of interventions for cerebral palsy: a systematic review.

AIM: Economic appraisal can help guide policy-making for purchasing decisions, and treatment and management algorithms for health interventions. We conducted a systematic review of economic studies in cerebral palsy (CP) to inform future research. METHOD: Economic studies published since 1970 were identified from seven databases. Two reviewers independently screened abstracts and extracted data following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Any discrepancies were resolved by discussion. RESULTS: Of 980 identified references, 115 were included for full-text assessment. Thirteen articles met standard criteria for a full economic evaluation, two as partial economic evaluations, and 18 as cost studies. Six were full economic evaluations alongside clinical studies or randomized controlled trials, whereas seven involved modelling simulations. The economic case for administration of magnesium sulfate for imminent preterm birth is compelling, achieving both health gain and cost savings. Current literature suggests intrathecal baclofen therapy and botulinum toxin injection are cost-effective, but stronger evidence for long-term effects is needed. Lifestyle and web-based interventions are inexpensive, but broader measurement of outcomes is required. INTERPRETATION: Prevention of CP would avoid significant economic burden. Some treatments and interventions have been shown to be cost-effective, although stronger evidence of clinical effectiveness is needed. What this paper adds Cost-effectiveness evidence shows prevention is the most significant strategy. Some treatments are cost-effective, but stronger evidence for long-term effectiveness is required. Comparison of treatment costs is challenging owing to variations in methodologies and varying clinical indications.

The National Disability Insurance Scheme: a time for real change in Australia.

In Australia, the supports and services for persons with disabilities have long been underfunded and fragmented. Often, individuals did not receive the services they needed, but rather the services they were entitled to based on how or when they acquired their disability. As a result, there was an increasing reliance on ageing carers, a lack of permanent and respite accommodation, and reduced employment and educational opportunities. Individuals with disabilities and their families were often isolated and financially disadvantaged. In March 2013, legislation was passed in Australia to establish a National Disability Insurance Scheme, a radical new way of funding disability services. No longer would funding be directed to agencies, but rather to individuals who would make their own plan and select their preferred services and service providers, giving them more control over the services and supports they receive. The hope is that this change from a welfare-driven to an insurance-based model will improve equity of service delivery, levels of participation, and overall quality of life among Australians with disabilities and their families.

Activities and participation of children with cerebral palsy: parent perspectives.

PURPOSE: To explore parents' views of the activities and participation of children with cerebral palsy (CP) with a range of communicative abilities and the factors (personal and environmental) that influenced these. METHODS: Thirteen parents of children with CP aged 4-9 years participated in semi-structured individual interviews. Interviews were recorded, transcribed and analysed thematically. Identified codes and themes were mapped to the domains of the International Classification of Functioning, Disability and Health - Children and Youth Version (ICF-CY). RESULTS: Parents' responses reflected all ICF-CY domains comprising activity, participation and environmental factors. Codes were primarily mapped to the domains learning and applying knowledge, communication, mobility and interpersonal interactions and relationships. Key barriers identified included aspects of parents' own interactions with their child (e.g. not offering choices), unfamiliar people and settings, negative attitudes of others and children's frustration. Facilitators included support received from the child's family and school, being amongst children, having a familiar routine and the child's positive disposition. CONCLUSIONS: Despite the barriers experienced, children participated in a range of activities. Parents placed importance on communication and its influence on children's independence, behaviour and relationships. Barriers and facilitators identified highlight aspects of the environment that could be modified through intervention to enhance communication and participation. IMPLICATIONS FOR REHABILITATION: Children's activities and participation were largely related to early learning tasks (e.g. literacy), communication, mobility and interactions. Intervention aimed at improving activities and participation may address the various child, impairment, social and environment factors identified here as impacting on activities and participation (e.g. the child's personal characteristics, communication and physical impairments, the support and attitudes of others and the familiarity of the environment). Therapists will need to consider (and manage) the potential negative impact communication deficits may have on children's behaviour, independence and social skills which may in turn detrimentally impact on activity and participation.

Quality of life of adolescents with cerebral palsy: perspectives of adolescents and parents.

Quality of life (QOL) has emerged over the past 20 years as an outcome for measuring the effectiveness of health-improvement interventions. The Cerebral Palsy Quality of Life Questionnaire for Children (CPQOL-Child) is well regarded and now integrated into research internationally. We describe the results of qualitative research, using grounded research in which we aimed to identify domains of QOL for adolescents with cerebral palsy (CP) from adolescent and parent perspectives to guide the development of an adolescent version. Seventeen adolescents (nine males, eight females) aged 13 to 18 years (mean=15.53 SD=1.74), with varying levels of impairment (Gross Motor Function Classification System levels I n=5, II n=2, III n=6, IV n=4, and V n=6) and their parents (n=23) participated in semi-structured interviews. Questions included: 'What do you think is important to your (child's) QOL?' and 'How does CP impact on your (child's) life?' Fifteen themes were identified, including domains related to health issues in adolescence, participation, education, specific CP-related issues (pain and discomfort, communication), family issues, practical issues (financial resources), and changes associated with adolescence (sexuality, independence). The composition of these QOL domains reflects the developmental concerns of adolescents with CP, adding to the views of children, and strongly supports adolescent participation in the development of self-reported well-being and QOL measures. Many of the domains are consistent with child reports of QOL and thus it appears feasible and valid to develop a measure that will be transferable across childhood and adolescence.

Perceived stress, perceived social support, and wellbeing among mothers of school-aged children with cerebral palsy.

BACKGROUND: The study considers how severity of disability, perceived stress, and perceived social support impact on the wellbeing of a group of mothers caring for school-aged children with cerebral palsy. METHOD: Forty-three mothers attending clinics at the Royal Children's Hospital in Melbourne, Australia were interviewed and completed the Profile of Adaptation to Life Scale, the Satisfaction with Life Scale, the Perceived Stress Scale, and the Multidimensional Scale of Perceived Social Support. RESULTS: The results confirmed that severity of disability was not significantly related to maternal wellbeing, but that perceived stress and perceived social support significantly predicted wellbeing, both independently and together. CONCLUSION: Social support was found to have a slight to moderate role in mediating the impact of stress on these mothers.