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The purpose of this article is to describe our clinical experiences in using the Gross Motor Function Measure (GMFM) to evaluate motor development in children with Down syndrome and to provide strategies we found helpful in enhancing a child's adherence to standardized testing. The issues discussed are: (1) strategies for test administration; (2) modifications in administration and scoring; (3) reliability of the GMFM using the modified administration and scoring procedures; and (4) applications of the GMFM for clinical practice. The strategies and recommendations address the particular characteristics of children with Down syndrome and allow for their progress to be monitored relative to other children with Down syndrome rather than to children without motor delays. Future studies validating the use of specific goal areas for the administration and scoring of the GMFM for children with Down syndrome are recommended. [ABSTRACT FROM AUTHOR]

Abstract Background Adolescents with cerebral palsy (CP) show a reduced physical activity (PA). Currently there are no interventions for adolescents with CP in this critical life phase that optimise and maintain the individuals' physical activity in the long term. To develop such a program it is important to fully understand the factors that influence physical activity behaviours in adolescents with CP. The aim of this study is to explore what makes it easy or hard for adolescents with CP to be and to become physically active. Methods/Design A qualitative research method is chosen to allow adolescents to voice their own opinion. Because we will investigate the lived experiences this study has a phenomenological approach. Thirty ambulatory and non-ambulatory adolescents (aged 10-18 years) with CP, classified as level I to IV on the Gross Motor Function Classification System and 30 parents of adolescents with CP will be invited to participate in one of the 6 focus groups or an individual interview. Therapists from all Children's Treatment Centres in Ontario, Canada, will be asked to fill in a survey. Focus groups will be audio- and videotaped and will approximately take 1.5 hours. The focus groups will be conducted by a facilitator and an assistant. In preparation of the focus groups, participants will fill in a demographic form with additional questions on physical activity. The information gathered from these questions and recent research on barriers and facilitators to physical activity will be used as a starting point for the content of the focus groups. Recordings of the focus groups will be transcribed and a content analysis approach will be used to code the transcripts. A preliminary summary of the coded data will be shared with the participants before themes will be refined. Discussion This study will help us gain insight and understanding of the participants' experiences and perspectives in PA, which can be of great importance when planning programs aimed at helping them to stay or to become physically active.

Background: Transition 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., Objective: This study aims to develop an internet-based mobile app intervention for patients with brain-based disabilities to improve transition in care readiness., Methods: The 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., Results: We 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., Conclusions: A 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 Registration: ClinicalTrials.gov NCT03852550; https://clinicaltrials.gov/study/NCT03852550., (©Ariane Marelli, Ronen Rozenblum, Clara Bolster-Foucault, Alicia Via-Dufresne Ley, Noemie Maynard, Khush Amaria, Barb Galuppi, Sonya Strohm, Linda Nguyen, Claire Dawe-McCord, Connie Putterman, Adrienne H Kovacs, Jan Willem Gorter. Originally published in JMIR Pediatrics and Parenting (https://pediatrics.jmir.org), 01.10.2024.)

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., Competing Interests: JWG and JM received research grants from the Canadian Institutes of Health Research Strategy for Patient-Oriented Research, and JWG held the Scotiabank Chair in Child Health Research during the duration of this study. BG, LN, SS, and AV-DL were paid for their work as project staff members. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest., (© 2023 Nguyen, Dawe-McCord, Frost, Arafeh, Chambers, Arafeh, Pozniak, Thomson, Mosel, Cardoso, Galuppi, Strohm, Via-Dufresne Ley, Cassidy, McCauley, Doucet, Alazem, Fournier, Marelli and Gorter.)

Background: Engaging patients and family members as partners in research studies has become a widespread practice in healthcare. However, relatively little has been documented about what happens after the research study ends. For example, is patient and family engagement embedded in the wider infrastructure of organizations, and if so how? What are the long-term effects of engaging parents on research teams on the culture of how research is conducted? This study seeks to address these two gaps by examining how a culture of family engagement has been built over time at CanChild Centre for Childhood Disability Research at McMaster University in Ontario, Canada., Methods: This study is based on ethnographic research methodology and combines elements of organizational ethnography, interviews, and collaborative auto-ethnography with parent partners, researchers, staff, and trainees., Results: Since the inception of CanChild Centre for Childhood Disability Research at McMaster University in 1989, parents have been involved in research studies. Over time, this involvement evolved from being consulted on research studies to undertaking decision-making roles as partners and most recently as co-principal investigators. A growing infrastructure fosters a community of engagement that goes beyond the individual research study, and often beyond CanChild. This infrastructure consists of training, knowledge mobilization and social networking. In addition, the "softer" building blocks of CanChild's culture of engagement are an openness to learning from others, a commitment to relationship building, and a drive to grow and improve. These values are espoused by the leadership and are instilled in the next generation of researchers to inform both research and clinical work. While some challenges should be acknowledged when researchers and family partners work together on research studies, we identify a number of strategies that we have used in our studies to foster authentic and meaningful family-researcher partnerships., Conclusion: Engaging patients and families as partners in research constitutes a culture shift in health research, whereby studies about patients and families are carried out with them. Developing a community of engagement that transcends an individual research study is a step towards creating a culture of research that is truly shaped by the people about whom the research is being done., (© 2021. The Author(s).)

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 Transition TM 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., Competing Interests: Competing interests: JWG and AJM received research grants from the Canadian Institutes of Health Research Strategy for Patient-Oriented Research. JWG holds the Scotiabank Chair in Child Health Research. AK and RR were paid in part for their work as consultants. BG, LN, SS, NM and AV-D-L were paid for their work as project staff members. LT was paid in part for his work as statistical consultant., (© Author(s) (or their employer(s)) 2021. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.)

Aim: To determine the stability of the Gross Motor Function Classification System (GMFCS), Manual Ability Classification System (MACS), and Communication Function Classification System (CFCS) over 1-year and 2-year intervals using a process for consensus classification between parents and therapists., Method: Participants were 664 children with cerebral palsy (CP), 18 months to 12 years of age, one of their parents, and 90 therapists. Consensus between parents and therapists on level of function was ≥92% for the GMFCS, MACS, and CFCS. A linearly weighted kappa coefficient of ≥0.75 was the criterion for stability., Results: Kappa coefficients varied from 0.76 to 0.88 for the GMFCS, 0.59 to 0.73 for the MACS, and 0.57 to 0.77 for the CFCS. For children younger than 4 years of age, level of function did not change for 58.2% on the GMFCS, 30.3% on the MACS, and 39.3% on the CFCS. For children 4 years of age or older, level of function did not change for 72.3% on the GMFCS, 49.1% on the MACS, and 55% on the CFCS., Interpretation: The findings support repeated classification of children over time. The kappa coefficients for the GMFCS are attributed to descriptions of levels for each age band. Consensus classification facilitates discussion between parents and professionals that has implications for shared decision-making., What This Paper Adds: The findings support repeated classification of children over time. Stability was higher for the Gross Motor Function Classification System than the Manual Ability Classification System and Communication Function Classification System. The function of younger children was more likely to be reclassified. Percentage agreement between parents and therapists using consensus classification varied from 92% to 97%. The intraclass correlation coefficient overestimated stability compared with the weighted kappa coefficient., (© 2018 Mac Keith Press.)

Purpose: To examine the relationship among the Gross Motor Function Classification System (GMFCS), the Manual Ability Classification System (MACS), and the Communication Function Classification System (CFCS) in children with cerebral palsy (CP) and to determine the average number and effect of health conditions., Methods: Participants were 671 children with CP aged 2 to 12 years from Canada and the United States. Cross-tabulation of functional classifications and averages were computed for the number and impact of health conditions and comparisons among groups., Results: A total of 78 of the 125 possible classification combinations were recorded. Most frequent were GMFCS I, MACS I, CFCS I; GMFCS I, MACS II, CFCS I; and GMFCS II, MACS II, CFCS I. With lower levels of function, the average number and average impact of associated health conditions increased., Conclusions: The use of functional profiles across classification systems, with data on the associated health conditions, provides a more comprehensive picture of CP than any single classification or measure.

Objectives: To assess use, utility and impact of transition interventions designed to support and empower self-management in youth with chronic health conditions during transition into adult healthcare., Design: A 4-year mixed-method prospective cohort study., Setting: 2 academic paediatric hospitals (13 clinics) in Canada., Participants: 50 adolescents (42% male; mean age 17.9±0.9 years; 20 underlying diagnoses) with transfer to adult care planned within 1 year., Interventions: The Youth KIT (an organisational tool that includes goal setting activities); an online transition mentor., Main Outcome Measures: Frequency of use, utility and impact of the transition interventions; goal achievement; post-transfer qualitative interviews with youth., Results: 50 participants were enrolled during their last year of paediatric care; 36 (72%) were followed into adult care. All participants had access to the transition interventions from enrolment until the end of the study (exposure time: 12-47 months). Most youth (85%) reported using the medical/health section of the Youth KIT at least once; 20 (40%) participants engaged in chats with the mentor. The overall perceived utility of both interventions was modest; the Youth KIT received the highest ratings for 'help with goal setting': (mean (SD): 4.2 (2.3)) on a 7-point Likert scale. 45 (90%) participants set 294 transition goals. Goal achievement performance and satisfaction increased over time (p≤0.001). The qualitative evidence revealed reasons behind the variability in use and utility of the interventions, the interconnectedness of life-course and healthcare transitions, and the need for stronger partnerships between paediatric and adult healthcare systems., Conclusions: Participants' perceptions about the utility of the Youth KIT and the online mentor were modest. Transition supports need to be carefully tailored, timed and integrated into healthcare systems. Individualised goal setting may be an important 'active ingredient' in optimising transition supports and outcomes. Interventions that focus on youth only are insufficient for empowering self-management., (Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://group.bmj.com/group/rights-licensing/permissions.)

Youth with cerebral palsy (CP) face significant barriers to participation in physical activity (PA). There is little information available about the nature of these barriers. Seventeen (17) youth and/or their parents participated in focus groups and individual interviews to identify factors that make it easy or hard to be physically active. Four themes emerged across functional levels: environmental and personal factors, limitations related to impairment in body structure and function, the perception that health benefits alone do not motivate youth to be physically active, and variable preferences for activity delivery. Dialogue with participants revealed that interventions to promote PA in youth should mitigate the interactions between personal and environmental factors that act as barriers to PA, and enhance the interactions that facilitate PA. Partnerships between researchers, policy makers, service providers, and families must be developed to address system barriers and build capacity in youth with CP and their communities.

Aim: To describe the development of context therapy, a new intervention approach designed for a randomized controlled trial., Method: Therapists were trained to change task and environmental factors to achieve parent-identified functional goals for children with cerebral palsy. Therapists did not provide any remediation strategies to change the abilities of the child. Theoretical constructs were developed using dynamic systems theory and the principles of family-centered care. A primary therapist model was used. A three-step intervention strategy was developed., Results: Therapists adhered to the treatment protocol. Parents participated in the development of both functional goals and intervention strategies., Interpretation: A therapy approach focusing on changing the task and the environment rather than children's impairments can be a viable treatment strategy and merits further investigation. The detailed description of the context therapy approach allows replication by both researchers and clinicians. Such intervention descriptions are an important methodological consideration in rehabilitation research., (© The Authors. Developmental Medicine & Child Neurology © 2011 Mac Keith Press.)

Aim: This study evaluated the efficacy of a child-focused versus context-focused intervention in improving performance of functional tasks and mobility in young children with cerebral palsy., Method: A randomized controlled trial cluster research design enrolled 128 children (49 females, 79 males; age range 12 mo to 5 y 11 mo; mean age 3 y 6 mo, SD -1 y 5 mo) who were diagnosed with cerebral palsy. Children across levels I to V on the Gross Motor Classification System (GMFCS) were included in the study. Children were excluded if there were planned surgical or medication changes during the intervention period. Therapists from 19 children's rehabilitation centers were block randomized (by occupational therapist or physical therapist) to a treatment arm. Children from consenting families followed their therapists into their assigned group. Children received child-focused (n=71) or context-focused intervention (n=57) over 6 months, returning to their regular therapy schedule and approach between 6 and 9 months. The primary outcome measure was the Pediatric Evaluation of Disability Inventory (PEDI). Secondary outcome measures included the Gross Motor Function Measure (GMFM-66), range of motion of hip abduction, popliteal angle and ankle dorsiflexion, the Assessment of Preschool Children's Participation (APCP), and the Family Empowerment Scale (FES). Outcome evaluators were masked to group assignment and completed assessments at baseline, 6 months, and 9 months., Results: Ten children did not complete the full intervention, six in the child group and four in context group. GMFCS levels for children in the study were level I (n=37), level II (n=23), level III (n=21), level IV (n=21), and level V (n=26). There were no significant differences at baseline between the treatment groups for GMFCS level, parental education, or parental income. For the PEDI, there was no significant difference between the treatment groups, except for a small effect (p<0.03) on the Caregiver Assistance Mobility subscale between baseline and 9 months. The mean scores of both groups changed significantly on the Functional Skills Scales (p<0.001) and Caregiver Assistance Scales (p<0.02) of the PEDI after the 6-month intervention. There was no additional statistically significant change on the PEDI during the follow-up period from 6 to 9 months. A subgroup effect was found for age (p<0.001), with children younger than 3 years changing significantly more than older children. GMFCS level at baseline did not influence the amount of change on the PEDI scales. There were no significant differences between the treatment groups on the GMFM, range of motion measures, APCP or FES assessments. For the GMFM, there was a significant change over time from baseline to 6 months (p<0.001) and no significant change between 6 and 9 months. There was no adverse side effect as range of motion did not decrease in either group. Hip abduction increased significantly (p<0.01) at the 9-month assessment for both groups. For the APCP, significant changes for both treatment groups were found between baseline and 6 months for play intensity (p<0.04), physical activity intensity and diversity (p<0.001), and total score intensity (p<0.01)., Interpretation: This study shows that child- or context-focused therapy approaches are equally effective and that frequency of intervention may be a critical component of successful intervention. Further evaluation is required to identify the various 'dose-response' relations of amount of treatment and changes in functional abilities., (© The Authors. Developmental Medicine & Child Neurology © 2011 Mac Keith Press.)

Aim: To explore associations between clinical variables and decline in motor capacity in adolescents with cerebral palsy (CP)., Method: Participants included 76 males and 59 females, whose mean age at the beginning of the study was 14 years 6 months (SD 2.4, range 11.6-17.9); 51 at Gross Motor Function Classification System (GMFCS) level III, 47 at level IV, and 37 at level V. Ninety-six participants had tetraplegia, 32 had diplegia, and one had hemiplegia. Types of motor disorder were spastic n=98; mixed, n=11; dystonic, n=9; hypotonic, n=7; and ataxic n=3 (seven participants were not classified). Reliable raters collected data annually for 4 years on anthropometric characteristics, the Spinal Alignment and Range of Motion Measure, as well as the Gross Motor Function Measure, 66 items (GMFM-66); participants or their parents reported on health status (using the Health Utilities Questionnaire), pain, and exercise participation (using measures developed for this study). The predicted drop in GMFM-66 scores after childhood was calculated using data on the same children from an earlier study. Correlations were calculated between the drop in GMFM-66 scores and the average and change scores of the clinical variables (the alpha level for statistical significance of this exploratory study was 0.10)., Results: The drop in GMFM-66 score was significantly correlated with limitations in range of motion (r=0.42) and spinal alignment (r=0.28), and pain (r=0.16). Increases in triceps skinfold (r=-0.19), mid-arm circumference (r=-0.23), and the ratio of mid-arm circumference to knee height (r=-0.23) were associated with less decline., Interpretation: Preventing range-of-motion limitations and pain experiences and optimizing nutrition might contribute to less decline in the gross motor capacity of adolescents with CP. Further investigation is required to clarify the role other factors that contribute to maintained function over time.

In this study parents' systematic accounts of the health status of 408 school-aged children with cerebral palsy (CP) are reported (221 males, 187 females; mean age 8 years 5 months, SD 1 year 11 months; range 5 to 13 years), as are relations between severity of functional motor impairment and eight functional health status domains. Data were collected as part of a longitudinal study of the motor development of a population-based, stratified, random sample of children with CP from across Ontario, Canada. The Gross Motor Function Classification System (GMFCS) was used to classify severity of CP and functional health status was described with the eight-level Health Utilities Index-Mark 3. Rates of functional limitations in Mobility, Dexterity, Speech, and Vision were statistically significantly associated with GMFCS levels (all p<0.01), with correlation values (tau-b) of 0.82, 0.58, 0.46, and 0.36, respectively. Functional limitations in hearing (tau-b=0.16; p=0.04) and cognition (tau-b=0.27; p<0.01) were both statistically significantly associated with GMFCS levels, though correlations were low. Neither emotion (tau-b=0.03; p=0.24) nor pain (tau-b=0.07; p=0.37) was associated with degree of functional limitation as described by the GMFCS. Clinical and epidemiological implications of findings are discussed.

Objective: To create gross motor function growth curves for children with Down syndrome (DS) and to estimate the probability that motor functions are achieved by different ages., Design: Nonlinear growth curve analysis by using a 2-parameter (rate, upper limit) model., Setting: Early intervention programs, schools, and children's homes., Participants: One hundred twenty-one children with DS, ages 1 month to 6 years., Main Outcome Measures: Gross Motor Function Measure (GMFM) and severity of motor impairment., Results: The curves for children with mild (n = 51) and moderate/severe (n = 70) impairment were characterized by a greater increase in GMFM scores during infancy and smaller increases as the children approached the predicted maximum score of 85.9 or 87.9. The estimated probability that a child would roll by 6 months was 51%; sit by 12 months, 78%; crawl by 18 months, 34%; walk by 24 months, 40%; and run, walk up stairs, and jump by 5 years, 45% to 52%., Conclusions: Children with DS require more time to learn movements as movement complexity increases. Impairment severity affected the rate but not the upper limit of motor function. The results have implications for counseling parents, making decisions about motor interventions, and anticipating the time frame for achievement of motor functions.

The purpose of this article is to describe our clinical experiences in using the Gross Motor Function Measure (GMFM) to evaluate motor development in children with Down syndrome and to provide strategies we found helpful in enhancing a child's adherence to standardized testing. The issues discussed are: (1) strategies for test administration; (2) modifications in administration and scoring; (3) reliability of the GMFM using the modified administration and scoring procedures; and (4) applications of the GMFM for clinical practice. The strategies and recommendations address the particular characteristics of children with Down syndrome and allow for their progress to be monitored relative to other children with Down syndrome rather than to children without motor delays. Future studies validating the use of specific goal areas for the administration and scoring of the GMFM for children with Down syndrome are recommended.

Background and Purpose: Development of gross motor function in children with cerebral palsy (CP) has not been documented. The purposes of this study were to examine a model of gross motor function in children with CP and to apply the model to construct gross motor function curves for each of the 5 levels of the Gross Motor Function Classification System (GMFCS)., Subjects: A stratified sample of 586 children with CP, 1 to 12 years of age, who reside in Ontario, Canada, and are known to rehabilitation centers participated., Methods: Subjects were classified using the GMFCS, and gross motor function was measured with the Gross Motor Function Measure (GMFM). Four models were examined to construct curves that described the nonlinear relationship between age and gross motor function., Results: The model in which both the limit parameter (maximum GMFM score) and the rate parameter (rate at which the maximum GMFM score is approached) vary for each GMFCS level explained 83% of the variation in GMFM scores. The predicted maximum GMFM scores differed among the 5 curves (level I=96.8, level II=89.3, level III=61.3, level IV=36.1, and level V=12.9). The rate at which children at level II approached their maximum GMFM score was slower than the rates for levels I and III. The correlation between GMFCS levels and GMFM scores was (.91. Logistic regression, used to estimate the probability that children with CP are able to achieve gross motor milestones based on their GMFM total scores, suggests that distinctions between GMFCS levels are clinically meaningful., Conclusion and Discussion: Classification of children with CP based on functional abilities and limitations is predictive of gross motor function, whereas age alone is a poor predictor. Evaluation of gross motor function of children with CP by comparison with children of the same age and GMFCS level has implications for decision making and interpretation of intervention outcomes.