The Pediatric Autism Research Cohort (PARC) Study: protocol for a patient-oriented prospective study examining trajectories of functioning in children with autism.

INTRODUCTION: The developmentally variable nature of autism poses challenges in providing timely services tailored to a child's needs. Despite a recent focus on longitudinal research, priority-setting initiatives with stakeholders highlighted the importance of studying a child's day-to-day functioning and social determinants of health to inform clinical care. To address this, we are conducting a pragmatic multi-site, patient-oriented longitudinal investigation: the Pediatric Autism Research Cohort (PARC) Study. In young children (<7 years of age) newly diagnosed with autism, we will: (1) examine variability in trajectories of adaptive functioning from the point of diagnosis into transition to school; and (2) identify factors associated with trajectories of adaptive functioning. METHODS AND ANALYSIS: We aim to recruit 1300 children under 7 years of age with a recent (within 12 months) diagnosis of autism from seven sites: six in Canada; one in Israel. Participants will be followed prospectively from diagnosis to age 8 years, with assessments at 6-month intervals. Parents/caregivers will complete questionnaires administered via a customized online research portal. Following each assessment timepoint, families will receive a research summary report describing their child's progress on adaptive functioning and related domains. Analysis of the longitudinal data will map trajectories and examine child, family and service characteristics associated with chronogeneity (interindividual and intraindividual heterogeneity over time) and possible trajectory turning points around sensitive periods like the transition to school. ETHICS AND DISSEMINATION: Ethics approvals have been received by all sites. All parents/respondents will provide informed consent when enrolling in the study. Using an integrated knowledge translation approach, where stakeholders are directly engaged in the research process, the PARC Study will identify factors associated with trajectories of functioning in children with autism. Resulting evidence will be shared with government policy makers to inform provincial and national programs. Findings will be disseminated at conferences and published in peer-reviewed journals.

Functional difficulties in children and youth with autism spectrum disorder: analysis of the 2019 Canadian Health Survey on Children and Youth. / Difficultés fonctionnelles chez les enfants et les jeunes atteints d'un trouble du spectre de l'autisme : une analyse de l'Enquête canadienne sur la santé des enfants et des jeunes de 2019.

INTRODUCTION: This study examined the prevalence of functional difficulties and associated factors in Canadian children/youth aged 5 to 17 years diagnosed with autism spectrum disorder (ASD). METHODS: We analyzed data from the 2019 Canadian Health Survey on Children and Youth (CHSCY), a nationally representative survey of Canadian children/youth that used the Washington Group Short Set on Functioning (WG-SS) to evaluate functioning in six daily tasks. For each functional domain, binary outcomes were derived (no/some difficulty, a lot of difficulty/no ability). We used logistic regression to identify associations between demographic characteristics, educational experiences, and perceived mental and general health and the most common functional difficulties, namely those related to remembering/concentrating, communication and self-care. All estimates were weighted to be representative of the target population. The bootstrap method was used to calculate variance estimates. RESULTS: Analysis of the records of 660 children/youth with ASD revealed that the most common functional difficulties were remembering/concentrating (22%; 95% CI: 18-27), communicating (19%; 95% CI: 15-23) and self-care (13%; 95% CI: 10-17). Lower perceived mental health was associated with increased functional difficulties with remembering/concentrating. ASD diagnosis at a lower age and lower perceived general health were associated with increased functional difficulty with communication. Parental expectations for postsecondary education were associated with decreased functional difficulty for self-care. CONCLUSION: One or more functional difficulties from the WG-SS was present in 39% of Canadian children/youth aged 5 to 17 years with ASD. Functional difficulties with remembering/concentrating, communication and self-care were most common.

Establishing and sustaining authentic organizational partnerships in childhood disability research: lessons learned.

There is an increased interest from both researchers and knowledge users to partner in research to generate meaningful research ideas, implement research projects, and disseminate research findings. There is accumulating research evidence to suggest the benefits of engaging children/youth with disabilities and their parents/families in research partnerships; however, less is known about the benefits of, and challenges to, engaging organizations as partners in research. The purpose of this commentary is to reflect on successful organizational partnership experiences from the perspectives of researchers at an internationally-recognized childhood disability research centre (CanChild), and to identify and share key ingredients for developing partnerships between organizations and academic institutions. A companion study is underway to examine partnership experiences with CanChild from the partners' perspective. Four CanChild researchers and two co-facilitators participated in a collaborative auto-ethnography approach to share experiences with organizational research partnerships and to reflect, interpret, and synthesize common themes and lessons learned. The researchers and facilitators met virtually via Zoom for 105 min. Researchers were asked to discuss the following: the formation of their organizational partnerships; if/how partnerships evolved over time; if/how partnerships were sustained; and lessons learned about benefits and challenges to building research partnerships with organizations. The meeting was recorded, transcribed verbatim, and analyzed by the facilitators to identify and synthesize common experiences and reflections. Multiple rounds of asynchronous reflection and feedback supported refinement of the final set of analytic themes. Researchers agreed that partnerships with organizations should be formed through a mutual interest, and that partnerships evolved by branching to include new organizations and researchers, while also involving trainees. Researchers identified the importance of defining roles and responsibilities of key individuals within each partnering group to sustain the partnership. Lessons learned from organizational partnerships included reciprocity between the partnering organization and academic institution, leveraging small pockets of funds to sustain a partnership over time, and building a strong rapport with individuals in a partnership. This commentary summarized lessons-learned and provided recommendations for researchers and organizations to consider when forming, growing, and sustaining research partnerships over time.

Researchers and people who use research findings are partnering to create research projects and share results. There are examples of children with disabilities and their families participating in research partnerships, but less is known about the involvement of healthcare organizations and community organizations as research partners. The purpose of this article is to share successful examples of partnership between organizations and a childhood disability research centre from the perspective of researchers. Four researchers and two facilitators met to reflect on their experiences with organizational research partnerships. They met online for 105 min using Zoom software. The researchers were asked to talk about how their partnerships with organizations were formed, how they grew over time, and how they were maintained. The meeting was recorded, and the facilitators took the researchers' experiences and summarized them into common messages. Everyone then read the summary on their own and added their ideas. This happened three different times until everyone agreed on one set of ideas. The researchers agreed that partnerships with organizations should be formed through common goals, that they should grow to include new partners and junior researchers, and that clear roles and responsibilities were needed to keep the partnership going. The experiences shared in this article are valuable to other researchers and organizations that are interested in forming research partnerships.

24-hour movement guideline adherence and mental health: A cross-sectional study of emerging adults with chronic health conditions and disabilities.

BACKGROUND: Recent work has shown that individuals with chronic health conditions and disabilities (CCD) meet the 24-h movement guidelines at lower rates than population norms; however, the evidence base remains limited across different stages of the lifespan and very few studies have examined associations with mental health outcomes. OBJECTIVE: This study examined 24-h movement guideline adherence among emerging adults with CCD compared to those without and associations between guideline adherence and indicators of mental health. METHODS: This cross-sectional study used data from the 2020 cycle of the Canadian Campus Wellbeing Survey. A total of 17,874 emerging adults enrolled at 20 post-secondary institutions (mean age = 21.6 ± 2.94 years; 65.2% female), including 3336 who identified with a CCD, self-reported their movement behaviors (physical activity, sedentary behaviors, sleep) and completed measures of psychological distress and mental well-being. Logistic regressions models were computed to examine differences in guideline adherence. Propensity score weighted linear regression models were computed to examine associations between guideline adherence and indicators of mental health. RESULTS: Emerging adults with CCD had significantly lower odds of meeting the 24-h movement guidelines than their peers, and disparities in guideline adherence were most pronounced among those with multimorbidity, developmental, and physical disabilities. Guideline adherence was associated with significantly more favorable scores for psychological distress and mental well-being among those with and without CCD. CONCLUSIONS: Findings suggest emerging adults with CCD engage in less healthy movement behavior patterns than their peers, yet they appear to experience similar mental health benefits when they do meet the 24-h movement guidelines.

Physical Activity Trajectories in Early Childhood: Investigating Personal, Environmental, and Participation Factors.

INTRODUCTION/PURPOSE: To determine personal, environmental, and participation factors that predict children's physical activity (PA) trajectories from preschool through to school years. METHODS: Two hundred seventy-nine children (4.5 ± 0.9 yr, 52% boys) were included in this study. Physical activity was collected via accelerometry at six different timepoints over 6.3 ± 0.6 yr. Time-stable variables were collected at baseline and included child's sex and ethnicity. Time-dependent variables were collected at six timepoints (age, years) and included household income (CAD), parental total PA, parental influence on PA, and parent-reported child's quality of life, child's sleep, and child's amount of weekend outdoor PA. Group-based trajectory modeling was applied to identify trajectories of moderate-to-vigorous PA (MVPA) and total PA (TPA). Multivariable regression analysis identified personal, environmental, and participation factors associated with trajectory membership. RESULTS: Three trajectories were identified for each of MVPA and TPA. Group 3 in MVPA and TPA expressed the most PA over time, with increased activity from timepoints 1 to 3, and then declining from timepoints 4 to 6. For the group 3 MVPA trajectory, male sex (ß estimate, 3.437; P = 0.001) and quality of life (ß estimate, 0.513; P < 0.001) were the only significant correlates for group membership. For the group 3 TPA trajectory, male sex (ß estimate, 1.970; P = 0.035), greater household income (ß estimate, 94.615; P < 0.001), and greater parental total PA (ß estimate, 0.574; P = 0.023) increased the probability of belonging to this trajectory group. CONCLUSIONS: These findings suggest a need for interventions and public health campaigns to increase opportunities for PA engagement in girls starting in the early years. Policies and programs to address financial inequities, positive parental modeling, and improving quality of life are also warranted.

A core outcome set for multimorbidity risk in individuals with cerebral palsy.

AIM: To: (1) investigate the importance of outcome measurement instruments (OMIs) within a core outcome set (COS) for multimorbidity (at least two chronic health conditions) risk in individuals with cerebral palsy (CP); (2) investigate the feasibility of OMIs within the COS in international clinical research settings in adolescents and adults with CP; and (3) describe the associations between the COS data and Gross Motor Function Classification System (GMFCS) levels. METHOD: Eighty-three individuals with CP completed a survey on health outcomes: physical behaviour, nutrition, sleep, endurance, body composition, blood pressure, blood lipids, and glucose. A cross-sectional study assessed the feasibility of the COS in 67 adolescents and adults with CP (mean age 30y, SD 15y 1mo, min-max: 14-68y, 52.2% male) at four centres. Prevalence of multimorbidity risk and associations with GMFCS levels are described. RESULTS: Most participants rated physical behaviour, nutrition, sleep, and endurance as very important. Body composition, blood pressure, nutrition, and sleep were highly feasible since data were collected in 88% or more participants who consented to having the assessments. Physical behaviour, cardiorespiratory endurance, and blood draws were collected in less than 60% of participants. Total time sedentary (ρ=0.53, p<0.01) and endurance (ρ=-0.46, p<0.01) were significantly associated with GMFCS level. INTERPRETATION: The COS identified that most participants had poor sleep quality and endurance, did not have healthy diets, and showed increased sedentary behaviour. Individuals with CP valued these outcomes as most important, suggesting a need to assess these modifiable behaviours in this population. Objective measures of physical behaviour and cardiorespiratory endurance in the COS required additional personnel, time, and participant burden. We recommend that healthcare providers should perform a simpler first screen using questionnaire-based assessments and then focus the use of the remainder of the COS if required for the patient.

UM DESFECHO CENTRAL DEFINIDO PARA RISCO DE MULTIMORBIDADE EM INDIVÍDUOS COM PARALISIA CEREBRAL: OBJETIVO: (1) Investigar a importância dos instrumentos de medição de desfechos (IMD) dentro de um conjunto de desfechos principais (CDS) para risco de multimorbidade (pelo menos duas condições crônicas de saúde) em indivíduos com paralisia cerebral (PC); (2) Investigar a viabilidade de IMD dentro do CDS em cenários internacionais de pesquisa clínica em adolescentes e adultos com PC; e (3) Descrever as associações entre os dados do CDS e os níveis do Sistema de Classificação da Função Motora Grossa (GMFCS). MÉTODO: Oitenta e três indivíduos com PC completaram uma pesquisa sobre desfechos em saúde: comportamento físico, nutrição, sono, resistência, composição corporal, pressão arterial, lipídios no sangue e glicose. Um estudo transversal avaliou a viabilidade do COS em 67 adolescentes e adultos com PC (idade média de 30 anos, desvio padrão de 15 anos e 1 mês, min-max: 14-68 anos, 52,2% do sexo masculino) em quatro centros. São descritas a prevalência do risco de multimorbidade e as associações com os níveis de GMFCS. RESULTADOS: A maioria dos participantes classificou o comportamento físico, nutrição, sono e resistência como muito importantes. Composição corporal, pressão arterial, nutrição e sono foram altamente viáveis, uma vez que os dados foram coletados em 88% ou mais dos participantes que consentiram em realizar as avaliações. Comportamento físico, resistência cardiorrespiratória e coleta de sangue foram coletados em menos de 60% dos participantes. O tempo total de sedentarismo (ρ = 0,53, p < 0,01) e resistência (ρ = −0,46, p < 0,01) foram significativamente associados ao nível de GMFCS. INTERPRETAÇÃO: O CDS identificou que a maioria dos participantes tinha má qualidade e resistência do sono, não tinha dietas saudáveis e apresentava um comportamento sedentário aumentado. Indivíduos com PC valorizaram esses desfechos como mais importantes, sugerindo a necessidade de avaliar esses comportamentos modificáveis nessa população. Medidas objetivas de comportamento físico e resistência cardiorrespiratória no CDS exigiram pessoal adicional, tempo e sobrecarga do participante. Recomendamos que os profissionais de saúde realizem uma primeira triagem mais simples usando avaliações baseadas em questionários e, em seguida, concentrem o uso do restante do CDS, se necessário para o paciente.

Individual and family characteristics associated with health indicators at entry into multidisciplinary pediatric weight management: findings from the CANadian Pediatric Weight management Registry (CANPWR).

OBJECTIVES: (1) To explore individual and family characteristics related to anthropometric and cardiometabolic health indicators and (2) examine whether characteristics that correlate with cardiometabolic health indicators differ across severity of obesity at time of entry to Canadian pediatric weight management clinics. METHODS: We conducted a cross-sectional analysis of 2-17 year olds with overweight or obesity who registered in the CANadian Pediatric Weight Management Registry (CANPWR) between May 2013 and October 2017 prior to their first clinic visit. Individual modifiable health behaviors included dietary intake, physical activity, screen time, and sleep. Family characteristics included parental BMI, family medical history, socioeconomic status and family structure. Linear mixed effects stepwise regression analysis was performed to determine which characteristics were related to each health indicator: BMI z-score; waist circumference; waist to height ratio; blood pressure; glycemia; HDL cholesterol; non-HDL cholesterol; triglycerides. RESULTS: This study included 1296 children (mean age ± standard deviation: 12.1 ± 3.5 years; BMI z-score: 3.55 ± 1.29; 95.3% with obesity). Hours spent sleeping (estimated ß = -0.10; 95% CI [-0.15, -0.05], p = 0.0001), hours per week of organized physical activity (estimated ß = -0.32; 95% CI [-0.53, -0.11], p = 0.0026), daily sugared drink intake (estimated ß = 0.06; 95% CI [0.01, 0.10], p = 0.0136) and maternal BMI (estimated ß = 0.03; 95% CI [0.02, 0.04], p < 0.0001) were associated with BMI z-score (adj. R2 = 0.2084), independent of other individual and family characteristics. Physical activity, total sugared drink intake and sleep duration were associated with glycemia and non-HDL cholesterol, independent of child BMI z-score. However, irrespective of obesity severity, little of the variance (0.86-11.1%) in cardiometabolic health indicators was explained by individual modifiable health behaviors. CONCLUSIONS: Physical activity, total sugared drink intake and hours spent sleeping were related to anthropometric and some cardiometabolic health indicators in children entering pediatric weight management programs. This highlights the importance of these modifiable health behaviors on multiple health indicators in children with obesity.

Metabolically healthy obesity in children enrolled in the CANadian Pediatric Weight management Registry (CANPWR): An exploratory secondary analysis of baseline data.

Our study purpose was to determine the prevalence of metabolically healthy obesity (MHO) and examine factors associated with MHO in children with obesity. This cross-sectional study was a secondary, exploratory analysis of data that included 2-17 years old with a body mass index (BMI) ≥85th percentile from the CANadian Pediatric Weight management Registry. Children were classified as having MHO or metabolically unhealthy obesity (MUO) using consensus-based criteria. Those with MHO had normal triglycerides, high-density lipoprotein cholesterol, blood pressure, and fasting glucose. Logistic regression was used to examine factors associated with MHO, which included calculating odds ratios (ORs) and 95% confidence intervals (CIs). In total, 945 children were included (mean age: 12.3 years; 51% female). The prevalence of MHO was 31% (n = 297), with lower levels across increasing age categories (2-5 years [n = 18; 43%], 6-11 years [n = 127; 35%], 12-17 years [n = 152; 28%]). Children with MHO were younger, weighed less, and had lower BMI z-scores than their peers with MUO (all p < 0.01). MHO status was positively associated with physical activity (OR: 1.18; 95% CI: 1.01-1.38), skim milk intake (OR: 1.10; 95% CI: 1.01-1.19), and fruit intake (OR: 1.12; 95% CI: 1.01-1.24) and negatively associated with BMI z-score (OR: 0.69; 95% CI: 0.60-0.79), total screen time in hours (OR: 0.79; 96% CI: 0.68-0.92), and intake of fruit flavoured drinks (OR: 0.91; 95% CI: 0.84-0.99). These findings may help guide clinical decision-making regarding obesity management by focusing on children with MUO who are at relatively high cardiometabolic risk.

Implications of Disability Severity on 24-Hour Movement Guideline Adherence Among Children With Neurodevelopmental Disorders in the United States.

BACKGROUND: Research has established beneficial associations between 24-hour movement guideline adherence and several health outcomes in typically developing (TD) children, but these relationships are poorly understood in children with neurodevelopmental disorders (NDD). This study examined (1) 24-hour movement guideline adherence, (2) the influence of disability severity, and (3) associations between guideline adherence and health outcomes of TD children and children with NDD. METHODS: This cross-sectional study used data from the 2018 and 2019 cycles of the US National Survey of Children's Health. Parental/caregiver reports of movement behaviors (physical activity, screen time, and sleep), disability severity (limitations to daily activities), and health outcomes (general health status, anxiety, and depression) were provided for 8554 children with NDD and 19,669 TD children aged 6-17 years. RESULTS: Children with NDD had significantly lower odds of meeting each movement behavior guideline compared to TD children; these effects were most pronounced for those who experienced consistent limitations to daily activities. Meeting at least 2 guidelines significantly lowered the odds for anxiety and depression, and increased the odds for better general health for children with NDD. DISCUSSION: These findings suggest that degree of disability severity has a strong influence on adherence to 24-hour movement guidelines among children with NDD.

Blood pressure in adults with cerebral palsy: a systematic review and meta-analysis of individual participant data.

OBJECTIVES: This systematic review and meta-analysis was designed to determine the overall mean blood pressure and prevalence of hypertension among a representative sample of adults living with cerebral palsy by combining individual participant data. Additional objectives included estimating variations between subgroups and investigating potential risk factors for hypertension. METHODS: Potential datasets were identified by literature searches for studies published between January 2000 and November 2017 and by experts in the field. Samples of adults with cerebral palsy (n ≥ 10, age ≥ 18 years) were included if blood pressure data, cerebral palsy-related factors (e.g. cerebral palsy subtype), and sociodemographic variables (e.g. age, sex) were available. Hypertension was defined as at least 140/90 mmHg and/or use of antihypertensive medication. RESULTS: We included data from 11 international cohorts representing 444 adults with cerebral palsy [median (IQR) age of the sample was 29.0 (23.0-38.0); 51% men; 89% spastic type; Gross Motor Function Classification System levels I-V]. Overall mean SBP was 124.9 mmHg [95% confidence interval (CI) 121.7-128.1] and overall mean DBP was 79.9 mmHg (95% CI 77.2-82.5). Overall prevalence of hypertension was 28.7% (95% CI 18.8-39.8%). Subgroup analysis indicated higher blood pressure levels or higher prevalence of hypertension in adults with cerebral palsy above 40 years of age, men, those with spastic cerebral palsy or those who lived in Africa. BMI, resting heart rate and alcohol consumption were risk factors that were associated with blood pressure or hypertension. CONCLUSION: Our findings underscore the importance of clinical screening for blood pressure in individuals with cerebral palsy beginning in young adulthood.

Self-reported factors contributing to fatigue and its management in adolescents and adults with cerebral palsy.

PURPOSE: To explore the self-reported factors that generate fatigue and to describe fatigue self-management strategies from the perspectives of adolescents and adults with cerebral palsy (CP). MATERIALS AND METHODS: Text responses to open-ended questions of the Fatigue Impact and Severity Self-Assessment from 160 participants (mean age 22.4 years) across all GMFCS levels were coded using inductive line-by-line coding and then grouped together to generate larger categories for each question. Frequency counts associated with each category were then summarized descriptively by Gross Motor Function Classification System level. RESULTS: The most commonly reported contributors to fatigue included the following: activity-related factors, general demands of life, sleep/rest, general health concerns, CP-related factors, mental health concerns, and environmental factors. The top five strategies participants reported to manage fatigue included rest or relaxation, sleeping or napping, changing or limiting their activities, being physically active, or using specific adaptations or assistive devices. CONCLUSIONS: Results from this study suggest that there are potentially modifiable factors, including activity level and sleep, that significantly contribute to fatigue for persons with CP; these could form the basis of interventions targeted at the prevention and management of fatigue.Implications for RehabilitationAs individuals with cerebral palsy who are physically active experience significant fatigue, clinicians need to address fatigue to enable these individuals to reap the health benefits of physical activity.Providing education and support to integrate self-management techniques, such as planning and pacing, may be an effective long-term strategy to support individuals to complete highly valued tasks.Interventions targeting modifiable fatigue-generating factors such as activity level, sleep, and mental health concerns are needed.

The Formula for Health and Well-Being in Individuals With Cerebral Palsy: Cross-Sectional Data on Physical Activity, Sleep, and Nutrition.

OBJECTIVE: To determine physical activity, sleep, and nutrition patterns in individuals with cerebral palsy (CP) and investigate the association of Gross Motor Function Classification System (GMFCS) and age with these health behaviors. METHODS: A cross-sectional study was conducted in an outpatient setting. Participants included adolescents and adults with CP (n=28; GMFCS level I-V; mean age 35.1±14.4 years). An Exercise Questionnaire or Leisure Time Physical Activity Questionnaire was used to measure physical activity in adolescents and adults, respectively. Sleep quality was measured using the Pittsburgh Sleep Quality Index (PSQI). An adapted version of the PrimeScreen questionnaire was used to assess nutrition. Linear regression analyses were performed to investigate the association between GMFCS and age with physical activity, sleep, and nutrition. RESULTS: The average total physical activity was 29.2±30.0 min/day. Seventy-five percent of the participants had poor sleep quality (PSQI score >5). Seventy-one percent reported "fair" eating behaviors; none reported "excellent" eating behaviors. Neither GMFCS nor age were significantly associated with PSQI score, PrimeScreen score, or total physical activity. A negative correlation existed between sleep quantity (hr/night) and PSQI score (r=-0.66, p=0.01). CONCLUSION: The triad of health components, consisting of physical activity, sleep, and nutrition, was not associated with GMFCS or age in our sample of 28 individuals with CP, suggesting that these three health behaviors should be assessed during clinical encounters of CP in adolescents and adults at all levels of the GMFCS.

Childhood Obesity and Cardiovascular Disease Risk: Working Toward Solutions.

The prevalence of obesity in childhood is high and continues to increase globally. It is currently estimated that 381 million children worldwide have overweight or obesity. This disease stems from multiple complex pathways that can present early in life. This is particularly concerning because childhood obesity is associated with cardiovascular risk factors that can lead to early atherosclerosis and premature cardiovascular disease (CVD). Hypertension, dysglycemia, dyslipidemia, and systemic inflammation are associated with vascular changes in childhood, and these contribute to increased risk of cardiovascular events in adulthood if not adequately treated. Interventions to treat childhood obesity include multicomponent family-based behaviour modification programs, which have been demonstrated to have moderate short-term effects on weight-related outcomes; their effects on cardiovascular risk factors, however, are less well understood. Although supervised, structured exercise interventions result in improvements in blood pressure, inflammation, carotid artery intima media thickness, dysglycemia, dyslipidemia, and endothelial dysfunction in children with obesity in the short term, our understanding of how to translate these interventions into long-term sustainable exercise or physical activity recommendations remains uncertain. Research focus in these areas will help in treating childhood obesity and associated CVD risk factors to prevent CVD development in adulthood.

The effects of an individualized health-risk report intervention on changes in perceived inactivity-related disease risk in adults with cerebral palsy.

BACKGROUND: To explore the effect of an individualized health-risk report intervention on changes in perceived disease risk in adults with cerebral palsy (CP). HYPOTHESES: 1. Perceived disease risk from those at risk for heart disease, obesity, and/or diabetes will be greater compared to those not at risk following receipt of health-risk information; 2. There will be a positive relationship between risk measures of inactivity-related diseases and post-intervention perceived disease risk; and increases in perceived disease risk will predict increases in physical activity (PA). METHODS: An individualized health-risk report was delivered to each participant. Three brief telephone interviews were conducted over two weeks to measure perceptions of disease risk and PA. Two-way repeated measures ANOVA and regression analyses were performed to address the study objectives. RESULTS: Thirty-one adults with CP (Gross Motor Function Classification System level I-V; age, 33.7 ±â€¯13.1y) completed the study. The health-risk report did not change the perceived risk of any inactivity-related disease. We found significant main effects for hypertension (p = 0.02; 95% CI [-1.1, -0.5]) on perceived risk of heart disease, and for waist circumference (p < 0.01; 95% CI [-1.3, -0.3]) and BMI (p < 0.01; 95% CI [-1.3, -0.3]) on perceived risk of obesity. PA did not change following the intervention. CONCLUSIONS: An individualized health-risk report intervention did not change perceived risk of inactivity-related disease or change PA behaviour. Blood pressure, waist circumference and BMI were salient measures of health that affected perceived disease risk in adults with CP; these health variables should be assessed and managed through clinical encounters.

Focus on Risk Factors for Cardiometabolic Disease in Cerebral Palsy: Toward a Core Set of Outcome Measurement Instruments.

OBJECTIVE: To identify existing outcome measurement instruments (OMIs) assessing risk factors for cardiometabolic disease in adolescents and adults with cerebral palsy (CP) reported on in the literature or used in the field. DATA SOURCES: The COnsensus-based Standards for the selection of health Measurement Instruments database of systematic reviews and 4 electronic databases (Embase, MEDLINE/Ovid, MEDLINE/Pubmed, PsychINFO) were searched up to June 19, 2017, that yielded 2594 articles. Experts in the field were consulted to identify any additional OMIs. STUDY SELECTION: Two reviewers independently applied inclusion criteria to select eligible studies using or evaluating measurement properties of OMIs assessing 1 of 8 outcomes: cardiorespiratory endurance, body size, body composition, physical behavior, sleep, nutrition, blood pressure, and blood lipids and glucose. Studies with an experimental or observational design including ≥10 adolescents or adults with CP were included. DATA EXTRACTION: One reviewer extracted data that were summarized for study and sample characteristics, outcomes, OMIs used, and if applicable data on measurement properties. Two reviewers rated the methodological quality and the quality of the OMIs. Feasibility for clinical practice and research was rated by experts in the field. DATA SYNTHESIS: Ninety OMIs were identified from 56 included articles and by the experts. Seventy OMIs pertained to cardiorespiratory endurance, body size, body composition, and physical behavior, whereas only 5 were identified for sleep and nutrition. Overall synthesis revealed that there is moderate to poor evidence for good quality of OMIs in this population. Based on feasibility for clinical practice, experts agreed on a single OMI per outcome (and 2 for cardiorespiratory endurance) to be included in a core set. CONCLUSION: Despite the range of available OMIs to assess risk factors for cardiometabolic disease in adolescents and adults with CP, evidence of good quality is often lacking. Nonetheless, a preliminary core set of 9 OMIs was systematically developed.

Emerging evidence for accelerated ageing and cardiovascular disease in individuals with cerebral palsy.

OBJECTIVE: To examine longitudinal changes in traditional and non-traditional risk factors for cardiovascular disease in individuals with cerebral palsy and to investigate relationships between age, Gross Motor Function Classification System (GMFCS) and risk of cardiovascular disease. METHODS: Individuals with cerebral palsy (n = 28 of 53 eligible participants; GMFCS levels I-V; follow-up mean age 35.1 years (standard deviation (SD) 14.4) participated in a longitudinal cohort study with 4.0 years (SD 1.2) follow-up. Traditional risk factors included waist circumference and systolic blood pressure. Non-traditional risk factors included carotid artery intima media thickness and distensibility, carotid-femoral pulse wave velocity, and flow-mediated dilation. RESULTS: Absolute (0.31 mm (SD 0.13) vs 0.22 mm (SD 0.08) , p = 0.045, 95% confidence interval (95% CI) 0.040, 0.151) and relative flow-mediated dilation (9.9 % (SD 4.7) vs 7.5 % (SD 2.6), p = 0.049, 95% CI 0.464, 4.42) decreased, while carotid artery intima media thickness (0.52 mm (SD 0.17) vs 0.67 mm (SD 0.33), p = 0.041, 95% CI -0.242, -0.074) increased from baseline to follow-up. No other risk factor changed significantly. Age at baseline was a significant independent predictor of carotid artery intima media thickness change (R-squared = 0.261, p = 0.031). CONCLUSION: Individuals with cerebral palsy experience significant changes in non-traditional risk factors for cardiovascular disease over 4 years, in the face of no changes in traditional risk factors. Compared with findings in the literature from the general population, these risk factors progress at a faster rate and at a younger age in individuals with cerebral palsy.

Multimorbidity risk assessment in adolescents and adults with cerebral palsy: a protocol for establishing a core outcome set for clinical research and practice.

BACKGROUND: Estimates of multimorbidity, defined as the presence of at least two chronic conditions, some of which attributable to modifiable behaviours, are high in adults with cerebral palsy (CP). An assessment protocol evaluating multimorbidity risk is needed in order to develop and evaluate effective interventions to optimize lifelong health in individuals with CP. The aim of this protocol paper is to describe the development of a core outcome set (COS) for assessing multimorbidity risk in adolescents and adults with CP, to be used in clinic and research. METHODS: The expert consortium will first define the target population and outcomes to be measured. Through a process of literature review and an international Delphi survey with expert clinicians and researchers, we will then determine which outcome measurement instruments (OMIs) can best measure those outcomes. The resulting OMIs will be used in a feasibility study with adolescents and adults with CP from an international clinical research network. Finally, a face-to-face stakeholder meeting with adolescents and adults with CP, their families/caregivers and researchers and clinicians who are experts in CP, will be organized to reach final agreement on the COS. DISCUSSION: This COS will guide clinicians and researchers in assessing multimorbidity risk in adolescents and adults with CP. The inclusion of experts and individuals with CP from international locations for establishing the COS lends strong support to its generalizability. Evidence of its feasibility and approval from all stakeholders will enable implementation in clinical practice, and guide future research using the COS in individuals with CP.

Cardiovascular disease and related risk factors in adults with cerebral palsy: a systematic review.

AIM: To summarize the literature on the prevalence of cardiovascular disease (CVD), risk factors of CVD, and CVD-related mortality in adults with cerebral palsy (CP). METHOD: A systematic review was conducted by searching the PubMed, Embase, MEDLINE (Ovid), Cochrane, and CINAHL databases. Selection criteria included adults with CP aged 18 years or over. Methodological quality was assessed using the Newcastle-Ottawa scale for observational studies. Data were reported descriptively. RESULTS: Nineteen studies met the inclusion criteria. Only one study reported directly on the presence of CVD in adults with CP, which found adults with CP reported greater CVD conditions than adults without CP (15.1 vs 9.1%, p<0.001). The most commonly reported risk factor of CVD in adults with CP was overweight/obesity. Five studies included data on CVD-related mortality in persons with CP, where CVD-related and circulatory system-related deaths were elevated and more common at a younger age in adults with CP than in the general population. INTERPRETATION: The prevalence of CVD and the risk of death because of CVD in this population seems increased, though the knowledge base is fragmented by studies that are small in size and geographically isolated. Further research is required to understand prevalence of risk factors among adults with CP, in particular overweight/obesity. WHAT THIS PAPER ADDS: Overweight and obesity are risk factors of cardiovascular disease (CVD) that are commonly reported in adults with cerebral palsy (CP). CVD-related and circulatory system-related deaths are elevated in individuals with CP compared to the general population.

ENFERMEDAD CARDIOVASCULAR Y FACTORES DE RIESGO RELACIONADOS EN ADULTOS CON PARÁLISIS CEREBRAL: UNA REVISIÓN SISTEMÁTICA: OBJETIVO: Análisis de la literatura publicada sobre la prevalencia de la enfermedad cardiovascular (ECV), los factores de riesgo de ECV y la mortalidad relacionada con ECV en adultos con parálisis cerebral (PC). MÉTODO: Se realizó una revisión sistemática buscando en las bases de datos de PubMed, Embase, Medline (Ovid), Cochrane y CINAHL. Los criterios de selección incluyeron adultos con PC de 18 años o más. La calidad metodológica se evaluó mediante la Escala de Newcastle Ottawa para estudios observacionales. Los datos se presentan descriptivamente. RESULTADOS: Diecinueve estudios cumplieron los criterios de inclusión. Solo un estudio informó directamente sobre la presencia de ECV en adultos con PC, el cual encontró mayor frecuencia de ECV en adultos con PC que en los adultos sin PC (15,1 % vs. a 9,1%, p <0,001). El sobrepeso / obesidad fue el factor de riesgo de ECV reportado más frecuentemente en adultos con PC. Cinco estudios que incluyeron datos sobre la mortalidad relacionada con ECV en personas con PC encontraron que el número de muertes relacionadas con ECV y con el sistema circulatorio fue más alto, y a una edad más temprana en adultos con PC que en la población general. INTERPRETACIÓN: La prevalencia de ECV y el riesgo de muerte por ECV está elevado en personas con PC. Sin embargo, el conocimiento sobre este tema es incompleto, compuesto por estudios de tamaño pequeño y geográficamente aislados. Se necesita más investigación para comprender la prevalencia de ECV y los factores de riesgo en adultos con PC, en particular sobrepeso / obesidad.

DOENÇA CARDIOVASCULAR E FATORES DE RISCO RELACIONADOS EM ADULTOS COM PARALISIA CEREBRAL: UMA REVISÃO SISTEMÁTICA: OBJETIVO: Sintetizar a literatura sobre a prevalência de doença cardiovascular (DCV), fatores de risco para DCV, e mortalidade relacionada a DCV em adultos com paralisia cerebral (PC). MÉTODO: Uma revisão sistemática foi conduzida por meio de pesquisa nas bases de PubMed, Embase, Medline (Ovid), Cochrane, e CINAHL. Critérios de seleção incluíram adultos com PC com idade a partir de 18 anos. A qualidade metodológica foi avaliada usando a Escala Newcastle Ottawa para estudos observacionais. Os dados foram reportados descritivamente. RESULTADOS: Dezenove estudos atenderam aos critérios de inclusão. Apenas um estudo reportou diretamente a presença de DCV em adultos com PC, e encontrou que adultos com PC relataram mais DCV do que adultos sem PC (15,1 vs 9,1%, p<0,001). O fator de risco mais comumente relatado em adultos com PC foi sobrepeso/obesidade. Cinco estudos incluíram dados sobre a mortalidade relativa a DCV em pessoas com PC, em que mortes relacionadas a DCV e ao sistema circulatório foram elevadas e mais comuns em idade mais jovem em adultos com PC do que na populacão em geral. INTERPRETAÇÃO: A prevalência de DCV e o risco de morte causada por DCV nesta população parecem ser aumentados, embora o conhecimento seja fragmentado em esudos que são pequenos e geograficamente isolados. Mais pesquisas são necessárias para compreender a prevalência de fatores de risco entre adultos com PC, em particular a obesidade/sobrepeso.

Fatigue in adults with cerebral palsy: A three-year follow-up study.

OBJECTIVES: To describe the course of fatigue over a 3-year follow-up period in adults with cerebral palsy and to investigate the association of known determinants of fatigue (i.e. demographic characteristics and/or body composition) with change in fatigue. METHODS: Forty-one adults with cerebral palsy from a previous study of fatigue were invited to participate in a follow-up study. Twenty-three adults with cerebral palsy (Gross Motor Function Classification System (GMFCS) levels I-V; mean age 38 years 2 months, standard deviation (SD) 14 years 1 month)) agreed to participate (convenience sample). Fatigue was measured with the Fatigue Impact and Severity Self-Assessment (FISSA, range 31-157) questionnaire. The course of fatigue is described at group, subgroup (GMFCS) and individual levels. RESULTS: The mean FISSA score for all participants was 84.0 (SD 27.7) at baseline and 91.7 (SD 26.7) at follow-up. Despite variations among individuals in the change of fatigue, there was no statistically significant difference in FISSA score over time (p = 0.087, 95% confidence interval (95% CI) -16.7 to 1.22). No known determinants of fatigue predictive of change in FISSA scores were found. DISCUSSION: Fatigue appears to be relatively stable within adults with cerebral palsy over time, with a variable presentation between individuals and across GMFCS levels. Care providers should monitor and discuss fatigue in young individuals with cerebral palsy in order to attenuate fatigue later in life.