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Abstract Background Poorly supported transitions from pediatric to adult healthcare can lead to negative health outcomes for youth and their families. To better understand the current landscape of healthcare transition care across Canada, the Canadian Health Hub in Transition (the “Transition Hub”, established in 2019) identified a need to: (1) describe programs and services supporting the transition from pediatric to adult healthcare across Canada; and (2) identify strengths, barriers, and gaps affecting the provision of transition services. Methods Our project included two iterative steps: a national survey followed by a qualitative descriptive study. Service providers were recruited from the Transition Hub and invited to complete the survey and participate in the qualitative study. The survey was used to collect program information (e.g., setting, clinical population, program components), and semi-structured interviews were used to explore providers’ perspectives on strengths, barriers, and gaps in transition services. Qualitative data were analyzed using the Framework Method. Results Fifty-one surveys were completed, describing 48 programs (22 pediatric, 19 bridging, and 7 adult) across 9 provinces. Almost half of the surveyed programs were in Ontario (44%) and most programs were based in hospital (65%) and outpatient settings (73%). There was wide variation in the ages served, with most programs focused on specific diagnostic groups. Qualitative findings from 23 interviews with service providers were organized into five topics: (1) measuring transition success; (2) program strengths; (3) barriers and gaps; (4) strategies for improvement; and (5) drivers for change. Conclusions While national transition guidelines exist in Canada, there is wide variation in the way young people and their families are supported. A national strategy, backed by local leadership, is essential for instigating system change toward sustainable and universally accessible support for healthcare transition in Canada.

Introduction While principles of neuroplasticity and motor learning emphasise the potential of high dosage of physical rehabilitation in children and adolescents with acquired brain injury (ABI) during the subacute phase, we lack empirical evidence to demonstrate its impact in terms of meaningful outcomes. Clinical research is needed to investigate adequate dosage of physical rehabilitation and its effects on outcomes with reliable and validated outcome measurements. In this study we will investigate the feasibility of a highly intensive physical rehabilitation intervention and outcomes with reliable and valid outcome measurements.Methods and analysis Our study with a prospective case-series design will evaluate (1) The safety and feasibility of a high-intensive physical rehabilitation programme to improve functioning and participation in children and adolescents with ABI, and (2) The feasibility of a core set of clinical outcome measures to assess recovery on relevant International Classification of Functioning, Disability, and Health domains during the subacute phase. We aim to include 10–15 children and adolescents aged 6–20 years with moderate-severe ABI during subacute rehabilitation in a specialised youth department for inpatient rehabilitation in The Netherlands. In the rehabilitation programme, we aim at a highly intensive approach of 3–5 hours physical rehabilitation per day during inpatient rehabilitation. Frequent and systematic assessment (baseline, each consecutive 6 weeks -if applicable- and at discharge) through a core set of outcome measures will provide insights into the degree of recovery of motor and cognitive functioning. We use descriptive and inferential statistics and thematic content analysis for analysis of the data.Ethics and dissemination The study has been approved by the Medical Ethics Research Committee of the University Medical Hospital, Utrecht (reference number: 23U-0628). We aim to disseminate our findings in peer-reviewed journals and present at national and international conferences.

Objective: To evaluate psychometrics of wearable devices measuring physical activity (PA) in ambulant children with gait abnormalities due to neuromuscular conditions. Data Sources: We searched PubMed, Embase, PsycINFO, CINAHL, and SPORTDiscus in March 2023. Study Selection: We included studies if (1) participants were ambulatory children (2-19y) with gait abnormalities, (2) reliability and validity were analyzed, and (3) peer-reviewed studies in the English language and full-text were available. We excluded studies of children with primarily visual conditions, behavioral diagnoses, or primarily cognitive disability. We performed independent screening and inclusion, data extraction, assessment of the data, and grading of results with 2 researchers. Data Extraction: Our report follows Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. We assessed methodological quality with Consensus-based Standards for the selection of health measurement instruments. We extracted data on reported reliability, measurement error, and validity. We performed meta-analyses for reliability and validity coefficient values. Data Synthesis: Of 6911 studies, we included 26 with 1064 participants for meta-analysis. Results showed that wearables measuring PA in children with abnormal gait have high to very high reliability (intraclass correlation coefficient [ICC]+, test-retest reliability=0.81; 95% confidence interval [CI], 0.74-0.89; I2=88.57%; ICC+, interdevice reliability=0.99; 95% CI, 0.98-0.99; I2=71.01%) and moderate to high validity in a standardized setting (r+, construct validity=0.63; 95% CI, 0.36-0.89; I2=99.97%; r+, criterion validity=0.68; 95% CI, 0.57-0.79; I2=98.70%; r+, criterion validity cutoff point based=0.69; 95% CI, 0.58-0.80; I2=87.02%). The methodological quality of all studies included in the meta-analysis was moderate. Conclusions: There was high to very high reliability and moderate to high validity for wearables measuring PA in children with abnormal gait, primarily due to neurological conditions. Clinicians should be aware that several moderating factors can influence an assessment.

Abstract Background Since 2011 when the Canadian Institutes of Health Research launched the Strategy for Patient Oriented Research, there has been a growing expectation to embed patient-oriented research (POR) in the health research community in Canada. To meet this expectation and build capacity for POR in the field of neurodevelopmental disability and child health, in 2017 researchers and family leaders at CanChild Centre for Childhood Disability Research, McMaster University partnered with Kids Brain Health Network and McMaster Continuing Education to develop and implement a 10-week online Family Engagement in Research (FER) Course. Main text From its inception, the FER Course has been delivered in partnership with family leaders and researchers. The FER Course is innovative in its co-learning and community building approach. The course is designed to bring family partners and researchers together to co-learn and connect, and to develop competency and confidence in both the theory and practice of family engagement in research. Coursework involves four live online group discussions, individual review of course materials, weekly group activities, and a final group project and presentation. Upon completion of the FER Course, graduates earn a McMaster University micro-credential. Conclusions To meet a need in building capacity in POR, a novel course in the field of neurodevelopmental disability and child health has been co-created and delivered. Over six years (2018–2023), the FER Course has trained more than 430 researchers and family partners across 20 countries. A unique outcome of the FER Course is that graduates expressed the wish to stay connected and continue to collaborate well beyond the course in turn creating an international FER Community Network that continues to evolve based on need. The FER Course is creating a growing international community of researchers, trainees, self-advocates, and family partners who are championing the implementation of meaningful engagement in neurodevelopmental disability and child health research and beyond. The course is internationally recognized with an established record of building capacity in POR. Its uptake, sustainability, and scalability to date has illustrated that training programs like the FER Course are necessary for building capacity and leadership in family engagement in research.

BackgroundTransition from pediatric to adult health care varies and is resource intensive. Patient-centered health information technology (HIT) interventions are increasingly being developed in partnership with patients. ObjectiveThis study aims to develop an internet-based mobile app intervention for patients with brain-based disabilities to improve transition in care readiness. MethodsThe app was designed for patients aged 15 to 17 years with brain-based disabilities having the ability to use a mobile app. A multidisciplinary team, an industry partner, and a patient and family advisory council was assembled. We hypothesized that existing tools could be migrated into the app to address education, empowerment, and navigation. We used cognitive learning theory to support chapters targeting transition in care skill sets. We used the agile iterative methodology to engage stakeholders. ResultsWe developed a novel MyREADY Transition HIT platform. An electronic mentor supported cognitive learning with messaging, quizzes, rewards, and videos. We used gaming to guide navigation through a fictitious health care city. Adapting existing tools was achieved by the patient and family advisory council requesting personalization. Our iterative design required time-consuming back-end technology management. Developing the platform took 24 months instead of our grant-approved 12 months, impacting the onset of the planned trial within the allotted budget. ConclusionsA novel patient-centered HIT platform to improve health care transition was successfully developed in partnership with patients and industry. Careful resource management was needed to achieve timely delivery of the end product, flagging the cautious planning required to deliver HIT tools in time for the much-needed trials informing their clinical application. Trial RegistrationClinicalTrials.gov NCT03852550; https://clinicaltrials.gov/study/NCT03852550

BackgroundThe transition from pediatric to adult care poses a significant health system–level challenge impeding the delivery of quality health services for youth with chronic health conditions. In Canada and globally, the transition to adult care is regarded as a top priority in adolescent health in need of readily applicable, adaptable, and relevant national metrics to evaluate and benchmark transition success across disease populations and clinical care settings. Unfortunately, existing literature fails to account for the lack of engagement from youth and caregivers in developing indicators, and its applicability across chronic conditions, primary care involvement, and health equity considerations. ObjectiveOur proposed study aims to establish a consensus-driven set of quality indicators for the transition to adult care that are universally applicable across physical, developmental, and mental health conditions, clinical care settings, and health jurisdictions. MethodsUsing an integrated knowledge translation (iKT) approach, a panel comprising youth, caregivers, interdisciplinary health care providers, and health system leaders will be established to collaborate with our research team to ensure that the study methodology, materials, and knowledge dissemination are suitable and reflect the perspectives of youth and their families. We will then conduct an iterative 3-round Online Modified Delphi (OMD) study (n=160) to (1) compare and contrast the perspectives of youth, caregivers, health care providers, and health system leaders on quality indicators for transition; and (2) prioritize a key set of quality indicators for transition applicable across disease populations that are the most important, useful, and feasible in the Canadian context. Using the RAND/UCLA Appropriateness Method (RAM) multistage analytic approach, data from each panel and stakeholder group will be examined separately and compared to establish a key set of indicators endorsed by both panels. ResultsThe study is funded by the Canadian Institutes of Health Research and Physicians Services Incorporated. ConclusionsThis study will produce quality indicators to evaluate and inform action equitably to improve transition from pediatric to adult care for youth and their families in Canada. International Registered Report Identifier (IRRID)PRR1-10.2196/60860

Abstract Background While patient and family engagement in research has become a widespread practice, meaningful and authentic engagement remains a challenge. In the READYorNot™ Brain-Based Disabilities Study, we developed the MyREADY Transition™ Brain-Based Disabilities App to promote education, empowerment, and navigation for the transition from pediatric to adult care among youth with brain-based disabilities, aged 15–17 years old. Our research team created a Patient and Family Advisory Council (PFAC) to engage adolescents, young adults, and parent caregivers as partners throughout our multi-year and multi-stage project. Main body This commentary, initiated and co-authored by members of our PFAC, researchers, staff, and a trainee, describes how we corrected the course of our partnership in response to critical feedback from partners. We begin by highlighting an email testimonial from a young adult PFAC member, which constituted a “critical turning point,” that unveiled feelings of unclear expectations, lack of appreciation, and imbalanced relationships among PFAC members. As a team, we reflected on our partnership experiences and reviewed documentation of PFAC activities. This process allowed us to set three intentions to create a collective goal of authentic and meaningful engagement and to chart the course to get us there: (1) offering clarity and flexibility around participation; (2) valuing and acknowledging partners and their contributions; and (3) providing choice and leveraging individual interests and strengths. Our key recommendations include: (1) charting the course with a plan to guide our work; (2) learning the ropes by developing capacity for patient-oriented research; (3) all hands on deck by building a community of engagement; and (4) making course corrections and being prepared to weather the storms by remaining open to reflection, re-evaluation, and adjustment as necessary. Conclusions We share key recommendations and lessons learned from our experiences alongside examples from the literature to offer guidance for multi-stage research projects partnering with adolescents, young adults, and family partners. We hope that by sharing challenges and lessons learned, we can help advance patient and family engagement in research.

There is an increasing desire to study neurodevelopmental disorders (NDDs) together to understand commonalities to develop generic health promotion strategies and improve clinical treatment. Common data elements (CDEs) collected across studies involving children with NDDs afford an opportunity to answer clinically meaningful questions. We undertook a retrospective, secondary analysis of data pertaining to sleep in children with different NDDs collected through various research studies. The objective of this paper is to share lessons learned for data management, collation, and harmonization from a sleep study in children within and across NDDs from large, collaborative research networks in the Ontario Brain Institute (OBI). Three collaborative research networks contributed demographic data and data pertaining to sleep, internalizing symptoms, health-related quality of life, and severity of disorder for children with six different NDDs: autism spectrum disorder; attention deficit/hyperactivity disorder; obsessive compulsive disorder; intellectual disability; cerebral palsy; and epilepsy. Procedures for data harmonization, derivations, and merging were shared and examples pertaining to severity of disorder and sleep disturbances were described in detail. Important lessons emerged from data harmonizing procedures: prioritizing the collection of CDEs to ensure data completeness; ensuring unprocessed data are uploaded for harmonization in order to facilitate timely analytic procedures; the value of maintaining variable naming that is consistent with data dictionaries at time of project validation; and the value of regular meetings with the research networks to discuss and overcome challenges with data harmonization. Buy-in from all research networks involved at study inception and oversight from a centralized infrastructure (OBI) identified the importance of collaboration to collect CDEs and facilitate data harmonization to improve outcomes for children with NDDs.

Introduction Patient engagement and integrated knowledge translation (iKT) processes improve health outcomes and care experiences through meaningful partnerships in consensus-building initiatives and research. Consensus-building is essential for engaging a diverse group of experienced knowledge users in co-developing and supporting a solution where none readily exists or is less optimal. Patients and caregivers provide invaluable insights for building consensus in decision-making around healthcare, policy and research. However, despite emerging evidence, patient engagement remains sparse within consensus-building initiatives. Specifically, our research has identified a lack of opportunity for youth living with chronic health conditions and their caregivers to participate in developing consensus on indicators/benchmarks for transition into adult care. To bridge this gap and inform our consensus-building approach with youth/caregivers, this scoping review will synthesise the extent of the literature on patient and other knowledge user engagement in consensus-building healthcare initiatives.Methods and analysis Following the scoping review methodology from Joanna Briggs Institute, published literature will be searched in MEDLINE, EMBASE, CINAHL and PsycINFO databases from inception to July 2023. Grey literature will be hand-searched. Two independent reviewers will determine the eligibility of articles in a two-stage process, with disagreements resolved by a third reviewer. Included studies must be consensus-building studies within the healthcare context that involve patient engagement strategies. Data from eligible studies will be extracted and charted on a standardised form. Abstracted data will be analysed quantitatively and descriptively, according to specific consensus methodologies, and patient engagement models and/or strategies.Ethics and dissemination Ethics approval is not required for this scoping review protocol. The review process and findings will be shared with and informed by relevant knowledge users. Dissemination of findings will also include peer-reviewed publications and conference presentations. The results will offer new insights for supporting patient engagement in consensus-building healthcare initiatives.Protocol registration https://osf.io/beqjr

Abstract Background Authentic researcher-youth partnerships in patient-oriented research (POR) where the research responds to the needs expressed by youth themselves are essential to make research meaningful. While patient-oriented research (POR) is increasingly practiced, few training programs exist in Canada and none, to our knowledge, are tailored for youth with neurodevelopmental disabilities (NDD). Our primary objective was to explore the training needs of youth (ages 18–25) with NDD to enhance their knowledge, confidence, and skills as research partners. Our secondary objective was to identify the benefits and challenges of engaging youth with NDD in a POR approach. Methods Our team of four youth and one parent with lived experience [Youth Engagement in Research (YER) partners] and six researchers engaged in POR to investigate the primary objective via two phases: (1) individual interviews with youth living with NDD and (2) a two-day virtual symposium with focus groups with youth and researchers. Collaborative qualitative content analysis was employed to synthesize the data. Our secondary objective was assessed by asking our YER partners to complete the Public and Patient Engagement Evaluation Tool (PPEET) survey and participate in reflective discussions. Results Phase 1 participants (n = 7) identified various barriers and facilitators to their engagement in research and offered suggestions to meet their needs through minimizing barriers and integrating facilitators, which would subsequently enhance their knowledge, confidence, and skills as research partners. Informed by phase 1, phase 2 participants (n = 17) prioritized the following POR training needs: researcher-youth communication, research roles and responsibilities, and finding partnership opportunities. For delivery methods, participants stated the importance of youth representation, using Universal Design for Learning, and co-learning between youth and researchers. Based on the PPEET data and subsequent discussions, YER partners agreed that they were able to express views freely, feel that their views were heard, and that their participation made a meaningful difference. Challenges included scheduling difficulties, ensuring multiple methods for engagement, and working under short timelines. Conclusion This study identified important training needs for youth with NDD and for researchers to engage in meaningful POR, which can subsequently inform the co-production of accessible training opportunities with and for youth.

Objective: Adolescents with chronic rheumatic disease must increasingly take on more responsibility for disease management from parents as they transition from pediatric to adult care. Yet, there are limited resources to inform and support parents about transition. Here, we evaluate the impact of a Transition Toolkit, geared towards parents and adolescents, on transition readiness, and explore the potential impact of parent–adolescent communication. Methods: A prospective cohort study of youths aged 14–18 years old and their parents was performed. Participant demographics, disease characteristics, transition readiness scores (Transition-Q, max 100), and parent–adolescent communication scores (PACS, max 100) were collected at enrollment (when the Transition Toolkit was shared with adolescents and their parents. Generalized estimating equation (GEE) analyses determined the influence of the Toolkit on transition readiness and explored the role of parent–adolescent communication quality. Subgroup analyses were conducted by sex. Results: A total of 21 patients were included; 19 completed one post-intervention Transition-Q and 16 completed two. Transition-Q scores increased over time and the rate of increase doubled after the Toolkit was shared (β = 7.8, p < 0.05, and β = 15.5, p < 0.05, respectively). Conclusion: Transition readiness improved at each follow-up, the greatest increase was seen after the Toolkit was shared. Parent–adolescent communication quality did not appear to impact changes in transition readiness.

Background: A growing population of youth with medical complexity (YMC) are entering adult health care, education, and social systems in which their needs have been largely neglected. To better support YMC and their families, an understanding of how they manage the challenges of transitioning to adult services is needed. The aim of this study was to examine how families of YMC adapt to challenges and opportunities posed by the youth’s transition to adulthood and transfer to adult services. Methods: In partnership with two parent co-researchers and underpinned by complex adaptive systems and the Life Course Health Development framework, a qualitative explanatory case study was conducted. Seventeen participants from 11 families of YMC (aged 16–30) living in Ontario were recruited. Data from 21 semi-structured interviews were analyzed using reflexive thematic analysis and further refined through theory-driven analysis. Supplementary documents shared by participants were analyzed using directed content analysis. Findings: Three overarching themes were generated. “Imagining, pursuing, and building a good future” describes families’ priorities and visions for the youth’s life as an adult. “Perils and obstacles of an imposed transition” examines challenges that families face in their pursuit of a good future. Lastly, “surviving the transition” describes how families are forced to advocate, make sacrifices, and persist in their efforts to adapt to transition. Conclusions: Pediatric providers should offer anticipatory guidance, partner with families in advocacy, and provide psychological support during transition. Education for adult and primary care providers should focus on developing professional competencies in the safe care of YMC, building capacity through clinical exposure, and creating culturally safe environments. Most importantly, YMC and their families need a model of care that can provide integrated, holistic, multidisciplinary care management across the lifespan.

AimsTo analyze needs and requirements of Pediatric Physical Therapists (PPTs), parents, children and adolescents with and without developmental disabilities in the future use of an activity monitor prototype (AM-p) in everyday clinical practice.MethodsQualitative exploratory study with a thematic analysis approach, based on Braun and Clarke's six steps. Codes derived from the analysis and central themes were collated, based on Fleuren et al.'s groupings of determinants.ResultsWe interviewed 25 PPTs, 12 parents, and 12 children and adolescents. Within four groupings of determinants, we found nine themes: 1) development of information materials; 2) application: output visualization and ease of use; 3) design; 4) relevance and acceptance; 5) shared decision-making; 6) compatibility in daily living; 7) finances, 8) time, and 9) legislation and regulations.ConclusionsEnd-users have similar basic needs, with individual fine-tuning to be addressed during further development of the AM-p. A child-friendly design, information material, and an easy-to-use application to read and interpret results, need to be developed. Efficient training for PPTs is important for the use of the AM-p and analysis of results. Communication between PPTs and children as well as parents enhances shared decision-making. We recommend involving diverse end-users to enable maximum customization of the AM-p.

There is a growing number of youth with healthcare needs such as disabilities or chronic health conditions who require lifelong care. In Canada, transfer to the adult healthcare system typically occurs at age 18 and is set by policy regardless of whether youth and their families are ready. When the transition to adult services is suboptimal, youth may experience detrimental gaps in healthcare resulting in increased visits to the emergency department and poor healthcare outcomes. Despite the critical need to support youth with disabilities and their families to transition to the adult healthcare system, there is limited legislation to ensure a successful transfer or to mandate transition preparation in Canada. This advocacy and policy planning work was conducted in partnership with the Patient and Family Advisory Council (PFAC) within the CHILD-BRIGHT READYorNot™ Brain-Based Disabilities (BBD) Project and the CHILD-BRIGHT Policy Hub. Together, we identified the need to synthesize and better understand existing policies about transition from pediatric to adult healthcare, and to recommend solutions to improve healthcare access and equity as Canadian youth with disabilities become adults. In this perspective paper, we will report on a dialogue with key informants and make recommendations for change in healthcare transition policies at the healthcare/community, provincial and/or territorial, and/or national levels.

Patient and family engagement has become a widely accepted approach in health care research. We recognize that research conducted in partnership with people with relevant lived experience can substantially improve the quality of that research and lead to meaningful outcomes. Despite the benefits of patient-researcher collaboration, research teams sometimes face challenges in answering the questions of how patient and family research partners should be compensated, due to the limited guidance and lack of infrastructure for acknowledging partner contributions. In this paper, we present some of the resources that might help teams to navigate conversations about compensation with their patient and family partners and report how existing resources can be leveraged to compensate patient and family partners fairly and appropriately. We also present some of our first-hand experiences with patient and family compensation and offer suggestions for research leaders, agencies, and organizations so that the health care stakeholders can collectively move toward more equitable recognition of patient and family partners in research.

Plain English summary In more and more projects, researchers and young people are working together in partnership; but there is little guidance about how to organize this partnership. In this paper, we share what partnerships in six projects from Canada, Netherlands, and United Kingdom looked like, so that others can be inspired. To do so, a researcher and a young partner from each project were asked to together: (1) describe their project, (2) summarize the practical details about the collaboration and (3) think about things that went well or could be improved. We found that all projects had the same beliefs important to partnerships, like having respect for each other. Young people could work on parts of the project they liked in a way that worked for them. They were supported by staff, could join meetings and were appreciated for their work. Clear communication during and in-between meetings was helpful. Youth were often asked about the role they wanted in the project. While there was often no formal training on how to do research, there were many opportunities to learn. We offer six recommendations to researchers and young people who want to partner together: (1) It is okay to not know what the partnership will look like and there is no single recipe of how to partner; (2) Take your time; (3) Discuss how the partnership is going; (4) Think about who is doing what and why; (5) Consider the diversity of young partners. We hope others will share their experiences.

Objective: To develop consensus among experts to guide physical rehabilitation in children and adolescents with acquired brain injury during the subacute phase. Design: International Delphi study. Methods: A 3-round online Delphi study was conducted with 11 international experts in rehabilitation for children and adolescents with acquired brain injury. The first round consisted of open-ended questions; the second and third round consisted of ranking 139 statements on a 5-point Likert scale. Results: The panel reached consensus on 116/139 statements. Consensus was reached on the importance of age, pre-injury developmental stage and the clinical presentation of the child when determining content and focus of physical rehabilitation. In addition, consensus was reached on the importance of participation-focused interventions, and involvement of family members in goal-setting and therapeutic activities. Although dosage was deemed very important, no consensus was reached for determination of dose-response variables to suit and influence the child’s needs. Conclusion: This study provides a framework for clinicians to design physical rehabilitation interventions in children with acquired brain injury in the sub-acute recovery phase. The promotion of physical activity in meaningful contexts and involvement of family members are considered as important components to optimize recovery.

ObjectiveThe objectives of this study were to determine rates of sleep disturbances in children with neurodevelopmental disorders (NDDs) within and across disorders and compared to typically developing (TD) children and to describe differences above and below the clinical cut-off for sleep disturbances. In addition, we explored the associations between demographic variables, severity of disorder, sleep disturbances, internalizing symptoms, and health-related quality of life (HRQOL) in children with NDDs.MethodWe conducted cross-sectional data analyses of an existing database with community-dwelling children with NDDs (n = 1438) and TD children (n = 140) aged 4–12 years. Parent-reported measures on sleep disturbances using the Children's Sleep Habits Questionnaire (CSHQ), internalizing symptoms using the Revised Children's Anxiety and Depression Scale, and HRQOL using the KINDL-R were assessed. Hierarchical linear regression examined the associations between demographic variables, severity of disorder, sleep disturbances, internalizing symptoms, and HRQOL in children with NDDs.ResultsChildren with NDDs (8.5 ± 2.1 years, 69.9% M) had significantly greater total sleep disturbance index (TSDI) than TD children [(8.6 ± 2.3 years, 60.0% M) (mean difference = 6.88 [95% CI 5.37, 8.40]; p < 0.001) (n = 838 NDDs (58.3%); n = 120 TD (86.7%)]. Children with severe NDDs reported significantly greater TSDI above the clinical cut-off (i.e., ≥41; CSHQ) than those with less severe NDDs (p < 0.001). Internalizing symptoms (β = −0.082 [95% CI −0.144, −0.019]; p = 0.011) and TSDI (β = −0.226 [95% CI −0.380, −0.073]; p = 0.004) were significantly associated with HRQOL in children with NDDs.ConclusionSurveillance and management of sleep and internalizing symptoms are needed to improve HRQOL in children with NDDs. Commonalities in sleep disturbances for children with NDDs support transdiagnostic interventions to treat sleep.

Plain English summary More and more patients and family members are getting involved in health research studies as partners. However we do not know much about what happens after the research study ends. This article looks at how parents have been involved in research studies at CanChild Centre for Childhood Disability Research at McMaster University in Ontario, Canada. CanChild researchers, staff, students and parents were asked about their experiences of working together on research studies. One of the researchers then pooled together all of these stories, shared them with everyone to get their feedback, and wrote the initial draft of this article. All the people interviewed were invited to read the article and to add their thoughts and opinions until everyone was satisfied with the final product. Our shared stories show that a lot has changed since CanChild was established in 1989. At first, researchers consulted with parents when they were doing a study. Now, many parents are partners and co-principal investigators on research studies. CanChild has also developed opportunities for parents and researchers to get training in patient-family engagement and to network with each other outside of research studies. Researchers, staff, students and parents talked about what makes research partnerships successful, including: being open to learning from each other; taking the time to get to know each other as people; and always trying to do better. They also shared some of the challenges that come up on research studies and suggested strategies for working through them.

Objective The transition from pediatric to adult rheumatology care represents a particularly vulnerable time for patients with juvenile idiopathic arthritis (JIA) and childhood‐onset systemic lupus erythematosus (cSLE). Improving self‐management skills is important in optimizing health care transition. The study’s objectives were to 1) examine variability in transition readiness of adolescents and young adults within and between different ages, sexes, and disease types; 2) determine the association between age and transition readiness; and 3) identify specific challenges to transition readiness for adolescents. Methods Over 1 year, patients 14 to 20 years of age with JIA or cSLE were recruited from pediatric transition and young adult clinics at a single academic institution. Participants completed the 14‐item Transition‐Q at a single time point. Total scores range from 0 to 100; higher scores indicate greater health care self‐management skills as a proxy for transition readiness. Descriptive statistics summarized patient characteristics and Transition‐Q scores for the population. Regression analyses determined the association between age, sex, and disease type and Transition‐Q score. Results Among 70 participants, 61 had JIA and 9 cSLE (mean disease duration 4.6 years). The mean (SD) total Transition‐Q score was 59.8 (14.9). Age was significantly associated with Transition‐Q score (standardized β = 0.372l P = 0.002). The most commonly reported challenges were seeing the physician alone (without parents), making one’s own appointments, picking up prescriptions, and independent transportation for appointments. Conclusion Transition readiness appears to increase with patient age. There is significant variability in Transition‐Q scores between patients of the same age, suggesting that an individualized approach to improving self‐management skills is necessary.

Abstract Background Cerebral Palsy (CP) is a group of disorders that affect the development of movement and posture. CP results from injuries to the immature brain during the prenatal, perinatal, or postnatal stage of development. Neuroimaging research in CP has focused on the structural changes of the brain during early development, but little is known about brain’s structural and functional changes during late adolescence and early adulthood, a period in time when individuals experience major changes as they transition into adulthood. The work reported here served as a feasibility study within a larger program of research (MyStory Study). We aimed to determine whether it would be feasible to scan and obtain good quality data without the use of sedation during a resting state condition for functional connectivity (FC) analyses in young adults with CP. Second, we aimed to identify the FC pattern(s) that are associated with depressive mood ratings, indices of pain and fatigue, and quality of life in this group. Methods Resting state functional images were collected from 9 young people with CP (18–29 years). We applied a stringent head motion correction and quality control methods following preprocessing. Results We were able to scan and obtain good quality data without the use of sedation from this group of young individuals with CP who demonstrated a range of gross motor ability. The functional connectivity networks of interest were identified in the data using standard seed regions. Our analyses further revealed that higher well-being scores were associated with higher levels of FC between the Medial Pre-Frontal Cortex and the right Lateral Parietal regions, which are implicated in prosocial and emotion regulations skills. The implications of this association are discussed. Conclusion The findings of the present study demonstrate that it is feasible to conduct resting state functional connectivity in young adults with CP with different gross motor abilities without the use of sedation. Our results also highlight a neural circuitry that is associated with the self-report of quality of life and emotion regulation. These findings identify these regions/circuitries as important seeds for further investigations into mental health and wellbeing in CP.

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.

Introduction Transition from paediatric to adult care is a complex process, which poses significant challenges for adolescents with chronic physical and mental illnesses. For many, transfer to adult care is associated with poor health and psychosocial outcomes. Quality indicators to evaluate transition to adult care are needed to benchmark and compare performance across conditions and health systems. This systematic review aims to identify quality indicators for successful transition to adult care which can be applied across chronic physical and/or mental illnesses.Methods Published literature will be searched using MEDLINE, Embase and CINHAL from earliest available date to July 2021. Grey literature will be searched using the Grey Matters tool. Using a set of inclusion/exclusion criteria, two independent reviewers will screen titles and abstracts, followed by full-text review. Disagreements will be resolved by a third reviewer. Study selection and data extraction will follow the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Protocols. Study appraisal will be completed using the Appraisal of Guidelines for Research and Evaluation for Quality Indicators instrument. Extracted quality indicators will be categorised into a conceptual framework.Ethics and dissemination Results from this review will offer novel insights into quality indicators that may be used to measure and evaluate transition success across conditions, which will be disseminated via a Canadian transition collaborative, workshops and peer-reviewed publication. Extracted quality indicators will be further prioritised in a Delphi study with patients, caregivers and providers. This is a critical step in developing a core set of metrics to evaluate transitions to adult care. Ethics approval is not required as this review will identify and synthesise findings from published literature.PROSPERO registration number CRD42020198030.

Despite the initial thrust of research aimed at understanding the impact of the COVID-19 pandemic on youth with physical illness and their parents, knowledge gaps in the literature remain, providing the impetus for additional investigation. This study described changes in psychological distress from prior to during the COVID-19 pandemic for parents and youth with physical illness, compared parent-proxy and youth self-reported perceptions of COVID-19-related psychosocial health, and modeled factors associated with psychological and psychosocial distress. There were 147 parent–youth dyads (2–16 years) from MY LIFE—a longitudinal study of youth with physical illness. The Kessler-6 (K6) measured psychological distress for the time before the COVID-19 lockdown (December 19 to March 20) and during the pandemic (December 20 to March 21) among parents and youth. COVID-19-related psychosocial health was measured using the CRISIS. Parents and youth reported increases in K6 scores (d = 0.62 and 0.38). Parent-proxy reports on the K6 were lower vs. youth self-reports prior to and during the pandemic (d = 0.63 for both). In contrast, parents reported lower proxy CRISIS scores for worries (d = 0.38) and effects of social restrictions (d = 0.52). Pandemic parent K6 scores were associated with age, combined in-person and online schooling for youth, COVID-19-related worries, and effects of social restrictions. For youth, only COVID-19-related worries and effects of social restrictions were associated with K6 scores. Parent worries were associated with youth sex, parental stress, family functioning, online and combination learning, and social restrictions. Parental depression and worries were associated with effects of social restrictions. Youth worries were associated with online and combination learning, and social restrictions. Youth disability, online learning, and worries were associated with effects of social restrictions. Few clinical factors are associated with COVID-19-related psychological and psychosocial distress. Instead, parent/family factors and youth learning environment have prominent roles in predicting outcomes and have implications for the health, education, and social services systems.

Background: As children and adolescents with a chronic health condition (CHC) age and transition to adulthood, many will increasingly assume responsibilities for the management of their healthcare. For individuals with CHCs, family members including siblings often provide significant and varied supports. There are a range of resources in Canada to support siblings of individuals with a CHC, but these resources are not synthesized and the extent to which they relate to healthcare management remains unclear.Purpose: The purpose of this document review was to identify, describe, and synthesize the types of resources currently available to provide general information and healthcare management information about how siblings can provide support to individuals with CHCs in Canada.Methods: Print and electronic resources were systematically identified and retrieved from the websites of organizations, treatment centers, and children's hospitals that are part of Children's Healthcare Canada. Each unique resource was treated as a text document. Documents that met the following inclusion criteria were included: addressed the topic of siblings of individuals with a CHC and written in English. Data were extracted from included documents and qualitative conventional content analysis was conducted. Throughout the process of this review, we partnered with a Sibling Youth Advisory Council.Results: The systematic search yielded 1,628 non-duplicate documents, of which 163 documents met the inclusion criteria. Of the total of 163 documents, they were delivered in the following formats: 17 (10%) general informational products (e.g., booklets, videos) about a CHC and sibling relationships, 39 about support programs and workshops (24%), 34 news articles (21%) that described the roles of siblings, and 6 (3%) healthcare management informational products (e.g., toolkit, tipsheets), 31 blogs (19%) and 39 interviews (24%) with parents and siblings. In the blogs and interviews, siblings and parents described how siblings developed knowledge and skills for healthcare management, as well as their role and identity over time.Significance: This study identified that there are limited resources available about healthcare management for siblings of CHC in Canada. Resources are needed to facilitate conversations in the family about the role of siblings with healthcare management of their sibling with a CHC.

Children with developmental language disorders (DLDs) may experience barriers to communicative participation. Communicative participation is defined as 'participation in life situations in which knowledge, information, ideas or feelings are exchanged'. Barriers experienced in communicative participation cannot be explained by language competence alone and are thought to be influenced by contextual factors. A better understanding of these factors will contribute to tailored speech and language therapy services for children with DLD. We conducted a focus group study with 13 speech and language therapists' (SLTs) to explore their perspectives on contextual (environmental and personal) factors in early childhood that are associated with communicative participation in children with DLD. The personal factor of child well-being, and the environmental factors of familial support and SLT service provision were developed through thematic analysis. The potential mediating role of these factors on communicative participation implies that it is important to address contextual barriers and facilitators in speech and language therapy services.

Abstract Background Accurate measurement of physical behaviour is paramount to better understand lifestyle, health, and functioning, particularly in adults with physical disability as they may be at higher risk of sedentary lifestyle and subsequent negative health consequences. This study aimed: 1) to evaluate the criterion validity of a novel and clinically applicable activity monitor (AM, Activ8), in the detection of body postures and movements in adults with spastic cerebral palsy (CP); and 2) to evaluate the extent that the AM’s positioning affects validity. Methods In this cross-sectional study, 14 ambulatory adults with CP [9 men; mean (SD) age, 35.4 (13.1) years] performed standardized activities while wearing three Activ8 monitors - frontolateral thigh (primary position), frontal thigh, and pant pocket - and being video recorded (criterion measure). AM activity output was compared to synchronized video recordings. Absolute (seconds) and relative [(video time–AM time)/mean time, %] time differences between methods were calculated. Relative time differences of

Abstract 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.

Introduction Siblings share a lifelong bond in their relationship, and they may choose to provide support to their brother or sister with a neurodisability. Previous reviews summarised programmes that only focused on the behavioural, emotional and psychological outcomes of the siblings. There is a need to synthesise existing evidence and enhance our understanding about programmes for siblings to acquire knowledge, develop skills and become empowered that can help them to provide support to their brother or sister with a neurodisability. The objective of this review is to identify and map the characteristics and outcomes of programmes designed to prepare siblings in their future roles to support their brother or sister with a neurodisability.Methods and analysis This review will be conducted using the Joanna Briggs Institute methodology for scoping reviews. An integrated knowledge translation approach will be used by partnering with the Sibling Youth Advisory Council comprised of siblings of individuals with a disability throughout all review phases. Databases to be searched include PsycINFO, Cumulative Index of Nursing and Allied Health Literature, Sociological Abstracts, Education Resources Information Center, EMBASE, Web of Science, MEDLINE (Ovid) and SPORTDiscus, from date of inception to November 2020. Studies of programmes designed for siblings of individuals with neurodisabilities, with no exclusion on the age of siblings or context, and published in English will be included. Extracted data will include details of programme structure and content, eligibility criteria and participants, context, study methods and outcomes. A summary of the results will be presented in a tabular form to provide an overview of the programmes with an accompanying narrative summary to address the research questions of this review.Dissemination Findings from this review will be shared using dissemination strategies in partnership with the Sibling Youth Advisory Council. We will share the findings with key stakeholders such as healthcare providers, researchers, and patient and family advocacy groups.

Introduction Youth with brain-based disabilities (BBDs), as well as their parents/caregivers, often feel ill-prepared for the transfer from paediatric to adult healthcare services. To address this pressing issue, we developed the MyREADY TransitionTM BBD App, a patient-facing e-health intervention. The primary aim of this randomised controlled trial (RCT) was to determine whether the App will result in greater transition readiness compared with usual care for youth with BBD. Secondary aims included exploring the contextual experiences of youth using the App, as well as the interactive processes of youth, their parents/caregivers and healthcare providers around use of the intervention.Methods and analysis We aimed to randomise 264 youth with BBD between 15 and 17 years of age, to receive existing services/usual care (control group) or to receive usual care along with the App (intervention group). Our recruitment strategy includes remote and virtual options in response to the current requirements for physical distancing due to the COVID-19 pandemic. We will use an embedded experimental model design which involves embedding a qualitative study within a RCT. The Transition Readiness Assessment Questionnaire will be administered as the primary outcome measure. Analysis of covariance will be used to compare change in the two groups on the primary outcome measure; analysis will be intention-to-treat. Interviews will be conducted with subsets of youth in the intervention group, as well as parents/caregivers and healthcare providers.Ethics and dissemination The study has been approved by the research ethics board of each participating site in four different regions in Canada. We will leverage our patient and family partnerships to find novel dissemination strategies. Study findings will be shared with the academic and stakeholder community, including dissemination of teaching and training tools through patient associations, and patient and family advocacy groups.Trial registration number NCT03852550.

Abstract Background ‘About My Child’ 19-item version (AMC-19) is a parent-report measure developed to assess the complexity of a child’s life due to biological, psychological, social and environmental issues, that can be completed in approximately 5 min. AMC measures two dimensions of complexity: parental concerns and impact on the child. This paper examines the psychometric properties and parent-reported utility of the AMC-19 for children with disabilities or special health care needs. Method Data were gathered from two Canadian studies at CanChild: the ‘AMC-19 Pilot’ study and the ‘Service Utilization and Outcomes (SUO)’ study. The AMC-19 Pilot study data allowed us to explore internal consistency and test-retest reliability, as well as parental responses to two open-ended questions on the utility of the AMC-19. The SUO study provided data for analyses of internal consistency and scale property validation with type of diagnosis and service needs. Results The test-retest ICC was r = 0.83 for concerns and r = 0.87 for impact. Cronbach’s alpha across both studies ranged from 0.80 to 0.90. Parents’ comments on the AMC-19’s utility indicated support for the AMC-19, in particular to identify therapy needs and goals. Conclusions The AMC-19 demonstrates strong psychometric properties supporting it as a valuable measure for describing the level of complexity among children with disabilities. We recommend using the AMC-19 in health services research and clinical settings to build dialogue between family and therapists due to its utility reported by parents.

Abstract Background In the management of hypertonicity in children with cerebral palsy (CP), goals should be clearly identified in order to evaluate the effectiveness of botulinum toxin A (BoNT-A) treatment, specifically in non-ambulatory children and adolescents, Gross Motor Function Classification System (GMFCS), level IV or V. A retrospective chart review (Mesterman et al., 2013) identified the need for the development of a set of specific and meaningful goals linked to the International Classification of Functioning, Disability and Health (ICF) for future goal setting and evaluation in this population. Our objective is to create an inventory of goals based on the ICF framework that captures the needs and values of families with children with CP. Methods This cross-sectional observational study recruited parents of twenty children and youths with CP in GMFCS levels IV or V (mean age 11.2 years, SD 4.3, 13 males) who were assessed for BoNT-A treatment at the Spasticity Management Clinic at McMaster Children’s Hospital (Hamilton, ON). A previous inventory of goals was developed by a group of experts at a national botulinum toxin conference held in January 2014 (Montreal, Canada). The inventory of goals was further refined by asking the parents to select goals from the inventory list that they would like their child to accomplish after receiving BoNT-A treatment, and asking healthcare professionals for clarity and phrasing of goals in the inventory list. Results All parents identified body structure and function goals, with more than 75% of parents selecting reduction in muscle tone and increased range of movements in the upper and lower extremities. More than 50% of parents identified activity goals related to ease of caregiving. Two activity goals and three participation goals were missing from the inventory. Participation goals were identified by less than 5% of parents. Conclusion The inventory may be a helpful tool to facilitate a discussion about goal setting between healthcare professionals and families in the context of BoNT-A treatment. A future study is needed to conduct qualitative interviews to better understand the information that families may require about setting goals during BoNT-A treatment and to evaluate the usefulness of the inventory.

Introduction Multimorbidity, the co-occurrence of a chronic physical condition and mental disorder, affects a substantial number of children and youth and can lead to compromised quality of life, hardship for families, and an increased burden on the healthcare system. We are conducting a study to document the course of mental disorder in children and youth diagnosed with a chronic physical condition; identify predictors of child and youth multimorbidity; examine whether the effects of these predictors are moderated by relevant psychosocial and biological factors; explore potential inflammatory and stress biomarkers that mediate the onset of child and youth multimorbidity; and, assess whether multimorbidity in children and youth alters patterns of mental health service use.Methods and analysis Multimorbidity in Children and Youth Across the Life-course (MY LIFE) is a prospective study. Two hundred and fifty children and youth aged 2–16 years diagnosed with a chronic physical condition along with one parent will be recruited from the outpatient clinics at a paediatric tertiary care centre. Data will be collected using a multi-informant, multimethod design at four time-points (at recruitment, and at 6, 12 and 24 months postrecruitment). Parents will provide reports for all children/youth. In addition, youth ≥10 years will self-report. Mental disorder will be assessed using structured interviews. On completion of data collection, participant-reported data will be linked to provincial health records to identify mental health services use. Multilevel analyses (survival, proportional hazard, structural equation modelling) will be used to address MY LIFE objectives.Ethics and dissemination This study has been approved by the University of Waterloo Human Research Ethics Board and the Hamilton Integrated Research Ethics Board. Findings will be disseminated to key stakeholders using a number of outlets (peer-reviewed publications and conferences, lay informational pamphlets, social media).

Physical inactivity in youth with cerebral palsy (CP) places them at increased risk of developing cardiovascular disease. The current study assessed indices of arterial health in adolescents with CP, classified as levels I-II of the Gross Motor Function Classification System (GMFCS) (n=11, age 13.2±2.1 yr), in comparison to age- and sex-matched controls (n=11, age 12.4±2.3 yr). Groups were similar in anthropometric measurements, resting blood pressures, and heart rates. There were no group differences in brachial flow-mediated dilation (11.1±7.8 versus 6.1±3.6), carotid intima-media thickness (0.42±0.04 versus 0.41±0.03 mm), and distensibility (0.008±0.002 versus 0.008±0.002 mmHg) or central (4.3±0.6 versus 4.1±0.9 m/s) and peripheral pulse wave velocity (7.1±1.7 versus 7.6±1.1 m/s); CP versus healthy controls, respectively. Vigorous intensity physical activity (PA) was lower in the CP group (CP: 38±80 min versus controls: 196±174 min); groups were similar in light and moderate intensity PA levels. Arterial health of ambulatory youth with CP is not different from a control group despite lower vigorous PA levels. Similar studies need to examine individuals with more pronounced mobility limitations (GMFCS level III–V).

Objective. To determine the feasibility of physical activity monitoring in adolescents with cerebral palsy (CP). Methods. A convenience sample of ambulatory and non-ambulatory adolescents (N=23; 17 males, 6 females; mean age 13.5 y, SD 2.6 y; Gross Motor Function Classification System (GMFCS) distribution: n=9 Level I, n=5 Level II, n=5 Level III, n=4 Level IV) was recruited. Physical activity (PA) was objectively assessed using the ActiGraph GT1M activity monitor. Discomfort or adverse effects of wearing the accelerometers were recorded by participants. Levels of physical activity were determined as total PA, light PA (LPA), moderate PA (MPA), moderate-to-vigorous (MVPA), and vigorous PA (VPA) using cut-points recently validated for CP. Results. Most participants showed little reluctance. Mean daily MVPA for all participants was 30.7 minutes (SD 30.3), which corresponded to 2.7 (SD 2.4) minutes of MVPA per hour or 4.5% (SD 3.9) of the total monitoring time. Total PA and MVPA were greatest in ambulatory youth (GMFCS levels I and II) compared with youth who use a walking aid or wheelchair (GMFCS levels III and IV) (P