Christy Brown's 'My Left Foot': An insider's insights into growing up with cerebral palsy.

IMPORTANCE: The field of childhood disability has undergone a sea-change in the past two decades. Remarkably, 70 years ago, the ideas now taking root were expressed with poignant clarity by Ireland's Christy Brown, providing lessons that were there to be learned, illustrating why 'My Left Foot' remains a singular contribution to the literature about child development and disability. OBJECTIVE OF THIS ESSAY: The World Health Organization's 2001 reconsideration of 'disability' (the International Classification of Functioning, Disability and Health or ICF) has spawned considerable uptake and adaptation of contemporary concepts, notably with the 'F-Words for Childhood Disability' (now the 'F-words for Child Development'). Published in 1954, Christy Brown's ground-breaking poetic autobiography 'My Left Foot' resonates today with messages that bring the ICF to life vividly and memorably. REVIEW: The author, a developmental paediatrician, has refracted the themes of 'My Left Foot' through an ICF lens to illustrate that concepts now considered modern have long been in plain sight, but sadly ignored. Christy Brown's first-person narrative animates ideas and messages for all who work in the field of childhood disability. FINDINGS: This essay is a personal reflection that draws together both contemporary 21st century concepts and ideas from the time that Christy Brown was a young author reporting his perspectives and perceptions on living with 'disability'. CONCLUSIONS AND RELEVANCE: The lessons Christy Brown generously shared 70 years ago should be heeded today. In the context of modern thinking and action regarding 'childhood disability', we need an approach to all we do that sees and respects children with 'disabilities' as whole people, that situates them in the context of family and community, that identifies and promotes their strengths and aspirations within both the health professional community and the community at large and enables them to 'become' and to 'belong'.

Patient perspectives in pediatric neurology: a critical shift in the paradigm of outcome measurement.

This review explores children's self-reported outcome measurements in pediatric neurology. We examine the following questions: (1) What is meant by patient-reported health, functioning, and quality of life outcomes? (2) How can patients express whether the interventions they receive do more good than harm? (3) Why and how should pediatric neurology patients help determine the outcomes of interest? (4) What tools and recommendations are available to evaluate the outcomes of interest? Applying patients' perspectives across the processes of evaluation of medical interventions has become an important expectation. These developments, consistent with current healthcare goals, coincide with the evolution of pediatric neurology into a sophisticated diagnostic-interventional field that aims to prolong survival, decrease impairments and symptoms, and improve patients' well-being - the recognized essential endpoints of interest in all medicine.

Quality of life in adolescents with epilepsy, cerebral palsy, and population norms.

AIM: To compare: (1) self- and proxy-reported quality of life (QoL) in adolescents with epilepsy, cerebral palsy (CP), both epilepsy and CP, and a representative general population sample; and (2) parental stress between parents of adolescents with epilepsy, CP, or both epilepsy and CP. METHOD: This was a cross-sectional observational study with 496 adolescents with epilepsy, 699 with CP, 192 with both CP and epilepsy, and 15 396 from the general population, assessed with the KIDSCREEN-52 and Parenting Stress Index (PSI). RESULTS: All KIDSCREEN-52 domains showed statistically significant differences across groups. The epilepsy population showed clinically better scores for 'school environment' than the general population (Cohen's d=0.62). Parents scored adolescents with CP lower than adolescents with epilepsy or general populations on 'physical health' (d=0.57, d=0.55) and 'social-support and peers' (d=0.82, d=0.91). Parents of adolescents with CP scored them lower than parents of the epilepsy group on 'autonomy' (d=0.62). Parents of adolescents with epilepsy scored them lower on 'mood and emotions' (d=0.52) and 'social acceptance' (d=0.66) than the general population. PSI scores were better for parents of adolescents with CP than for parents of adolescents with epilepsy (d=2.12, d=2.70, d=3.35, d=1.67). INTERPRETATION: Adolescents with epilepsy or CP self-report equal or better QoL than the general adolescent population, which should comfort families and allow clinicians to address parental concerns. WHAT THIS PAPER ADDS: Adolescents with epilepsy, with or without cerebral palsy (CP), self-reported better school environment than adolescents in the general population. Proxy quality of life (QoL) results showed clinically important differences across groups in 6 out of 10 domains of the KIDSCREEN-52. Proxy-reported results showed poorer QoL scores for adolescents with epilepsy or CP than the general population. Parental stress level was lower in parents of children with CP or both CP and epilepsy, than in those with only epilepsy.

Changes in Caregiver Health in the Years Surrounding the Birth of a Child With Health Problems: Administrative Data From British Columbia.

BACKGROUND: Caregivers of children with health problems (CHPs; usually mothers) experience more physical and psychological health problems than those of children without health problems (non-CHPs). Primarily cross-sectional and survey-driven, this literature has not yet explored whether these health differences existed before the birth of the CHPs, or are exacerbated postbirth. METHODS: Using linked administrative health data on all mother-child dyads for children born in the year 2000 in British Columbia, Canada, we examined maternal health before, during, and after the birth of CHPs, and compared it between mothers of CHPs and non-CHPs with piecewise growth curve modeling. RESULTS: Compared with mothers of non-CHPs, mothers of CHPs had more physician visits (8.09 vs. 11.07), more medication types (1.81 vs. 2.60), and were more likely to be diagnosed with selected health conditions (30.9% vs. 42.5%) 4 years before the birth of the child. Over the 7-year postbirth period, the health of the 2 groups of mothers further diverged: while mothers of CHPs showed increases on physician visits and types of medication, mothers of non-CHPs did not experience any changes in physician visits and had less steep increases for types of medication. CONCLUSIONS: Health issues associated with having a child with a health problem may begin well before the birth of the child, but also appear to be exacerbated postbirth. The health challenges of caregivers of CHPs may be multifactorial, involving both preexisting conditions and the stresses associated with caring for a child with health problems.

Can behavioral strategies increase physical activity and influence depressive symptoms and quality of life among children with epilepsy? Results of a randomized controlled trial.

PURPOSE: This study examined whether increasing physical activity (PA) through 6 months of behavioral counseling positively influenced depressive symptoms and quality of life (QoL) over 12 months among children with epilepsy (CWE). METHODS: A longitudinal multisite randomized controlled trial (RCT) was conducted with 8-14-year-old children with active epilepsy. Participants wore a pedometer to track daily PA and completed 3 measures at 4 time points to examine depressive symptoms and QoL. Stratified by site and activity level, participants were randomized to an intervention or control group. The 6-month intervention included 11 behavioral counseling sessions targeting self-regulation of PA. To assess the associations among PA, depression scores, and QoL, primary analysis involved mixed-effects models. RESULTS: We recruited 122 CWE, of whom 115 were randomized (Mage = 11 ±â€¯2; 50% female) and included in the analysis. The intervention did not increase PA in the treatment compared with the control group. No differences were found between groups over time during the subsequent 6 months, where PA decreased among all participants. Results did not show differences between the groups and over time for measures of depressive symptoms and QoL. SIGNIFICANCE: The intervention did not improve or sustain PA levels over 12 months. Both groups demonstrated declines in PA over one year, but there were no changes in depression scores or QoL. As most participants were already nearly reaching the Canadian average of step counts of children their age, with a baseline daily step count of over 9000, there may be a challenge for further increasing PA over a longer period.

Patient-reported quality of life and biopsychosocial health outcomes in pediatric epilepsy: An update for healthcare providers.

In the 21st century, clinicians are expected to listen to, and understand their patients' views about, their conditions and the effects that these conditions have on their functioning, values, life goals, and welfare. The goals of this review are as follows: (i) to inform, update, and guide clinicians caring for children with epilepsy about developments in the content and new methods of research on patient-reported outcomes, quality of life, and functioning; and (ii) to discuss the value of using these concepts to explore the impact of diverse interventions that are implemented in daily practice. Drawing on the literature and our program of research over the past two decades, we focus on our current understanding of a variety of health concepts and recently acquired knowledge about their significance for the lives of patients and their families. We discuss the advantages of measuring patient-reported outcomes that tell us what is important to patients. We advise on what characteristics to look for when choosing a patient-reported measure, and the relevance of these considerations. In addition, we address gaps in research knowledge and the causes of confusion that have limited their use in our daily clinical practice.

Child- and parent-reported quality of life trajectories in children with epilepsy: A prospective cohort study.

OBJECTIVE: To describe the developmental trajectories of quality of life (QoL) in a large cohort of children with epilepsy, and to assess the relative contribution of clinical, psychosocial, and sociodemographic variables on QoL trajectories. METHODS: Five assessments during a 28-month prospective cohort study were used to model trajectories of QoL. Participants were recruited with their parents from six Canadian tertiary centers. A convenience sample of 506 children aged 8-14 years with epilepsy and without intellectual disability or autism spectrum disorder were enrolled. A total of 894 children were eligible and 330 refused participation. Participating children were, on average, 11.4 years of age, and 49% were female. Nearly one third (32%) had partial seizures. At baseline, 479 and 503 child- and parent-reported questionnaires were completed. In total, 354 children (74%) and 366 parents (73%) completed the 28-month follow-up. QoL was measured using the child- and parent-reported version of the Childhood Epilepsy QoL scale (CHEQOL-25). RESULTS: Child-reported QoL was fitted best by a six-class model and parent-reported QoL by a five-class model. In both models, trajectories remained either stable or improved over 28 months. Of these children, 62% rated their QoL as high or moderately high, defined as at least one standard deviation above the average CHEQOL-25 score. Greater family, classmate, and peer social support, fewer symptoms of child and parent depression, and higher receptive vocabulary were identified as the most robust predictors of better QoL (all p < 0.001). SIGNIFICANCE: Most children with epilepsy and their parents reported relatively good QoL in this first joint self- and proxy-reported trajectory study. Findings confirm the heterogeneous QoL outcomes for children with epilepsy and the primary importance of psychosocial factors rather than seizure and AED-specific factors in influencing QoL. These predictors that are potentially amenable to change should now be the focus of specific intervention studies.

Reflections on Ethics and Humanity in Pediatric Neurology: the Value of Recognizing Ethical Issues in Common Clinical Practice.

Our goals in this reflection are to (i) identify the ethical dimensions inherent in any clinical encounter and (ii) bring to the forefront of our pediatric neurology practice the myriad of opportunities to explore and learn from these ethical questions. We highlight specifically Beauchamp and Childress's principles of biomedical ethics. We use the terms ethics in common clinical practice and an ethical lens to remind people of the ubiquity of ethical situations and the usefulness of using existing ethical principles to analyze and resolve difficult situations in clinical practice. We start with a few common situations with which many of us tend to struggle. We describe what we understand as ethics and how and why developments in technology, novel potential interventions, policies, and societal perspectives challenge us to think about and debate ethical issues. Individual patients are not a singular population; each patient has their own unique life situations, culture, goals, and expectations that need to be considered with a good dose of humanity and humility. We believe that using an ethical lens-by which we mean making an explicit effort to identify and consider these issues openly-will help us to achieve this goal in practice, education, and research.

Participation, both a means and an end: a conceptual analysis of processes and outcomes in childhood disability.

This review outlines a conceptual approach to inform research and practice aimed at supporting children whose lives are complicated by impairment and/or chronic medical conditions, and their families. 'Participation' in meaningful life activities should be an essential intervention goal, to meet the challenges of healthy growth and development, and to provide opportunities to help ensure that young people with impairments reach their full potential across their lifespan. Intervention activities and research can focus on participation as either an independent or dependent variable. The proposed framework and associated hypotheses are applicable to children and young people with a wide variety of conditions, and to their families. In taking a fresh 'non-categorical' perspective to health for children and young people, asking new questions, and exploring issues in innovative ways, we expect to learn lessons and to develop creative solutions that will ultimately benefit children with a wide variety of impairments and challenges, and their families, everywhere.

Life course health development of individuals with neurodevelopmental conditions.

The life course health development (LCHD) model by Halfon et al. conceptualizes health development occurring through person-environment transactions that enable well-being and participation in desired social roles throughout life, areas that have not received adequate attention in healthcare. The aim of this 'perspectives' paper is to apply the six core tenets of the LCHD model and the concept of health development trajectories to individuals with lifelong neurodevelopmental conditions. We share the perspective that modifiable aspects of the environment often restrict health development; we then advocate that children, beginning at a young age, should engage in 'real-world' experiences that prepare them for current and future social roles. LCHD encourages future planning from the outset, continuity of care between pediatric and adult systems, and coordination of services and supports. We believe LCHD can be transformative in enabling healthy living of individuals with neurodevelopmental conditions.

Quality of life in children with epilepsy: How does it compare with the quality of life in typical children and children with cerebral palsy?

Our objective was to compare the quality of life (QoL) of children with epilepsy to that of typical children and children with cerebral palsy (CP). We measured self- and proxy-reported QoL of children with epilepsy and contrasted that with data for typical children (European KIDSCREEN project) and children with CP (SPARCLE study). Children ages 8-12 years with epilepsy were recruited from six Canadian sites. Same-aged children with CP and children in the general population aged 8-11 years came from several European countries. All participants completed the KIDSCREEN-52 questionnaire. Our results showed no clinically important differences (>0.5 SD) between self-reported QoL in 345 children with epilepsy compared with 489 children with CP or 5950 children in the general population. However, parents reported clinically important differences between the epilepsy and the other groups in five KIDSCREEN-52 domains. Compared with the CP group, parents of children with epilepsy reported better QoL in physical well-being (Cohen d=0.81), social support (d=0.80), and autonomy (d=0.72). Parents reported poorer QoL in the domains of mood and emotions compared with both contrast groups (d=-0.72 and d=-0.53), and in the domain of bullying compared with the CP group (d=-0.51). Families should find comfort in the results, which indicate that children with epilepsy do not perceive any important differences in QoL compared with their typical peers. The comparisons of parental reports detect their group-specific observations and worries that need to be addressed by the health-care providers and may require specifically designed assessment batteries followed by appropriate interventions.

Developing a non-categorical measure of child health using administrative data.

BACKGROUND: Few studies have examined the potential of linked administrative data for research on child health. This analysis describes the application of a non-categorical survey-based tool, the Children with Special Health Care Needs (CSHCN) Screener, to administrative data. DATA AND METHODS: Five Screener items were applied to linked administrative health data from Population Data British Columbia. Hospital admissions and demographic and community characteristics for a cohort of children aged 6 to 10 in 2006 were examined to validate the use of these items. RESULTS: Overall, 17.5% of children were identified as CSHCN. An estimated 14% of children used more medical care and 5.2% had more functional limitations than is usual for children of the same age; 3.3% were prescribed long-term medication; 1.9% needed/received treatment or counselling; and 0.1% needed/received special therapy. Boys were more likely than girls to be identified as CSHCN. INTERPRETATION: With some limitations, the CSHCN Screener can be applied to Canadian administrative health data.

A Bioecological framework to evaluate communicative participation outcomes for preschoolers receiving speech-language therapy interventions in Ontario, Canada.

BACKGROUND: The Preschool Speech and Language Program (PSLP) in Ontario, Canada, is a publically funded intervention service for children from birth to 5 years with communication disorders. It has begun a population-level programme evaluation of children's communicative participation outcomes following therapy. Data are currently being collected for all children who access this service--over 50,000 children each year. AIMS: To understand and evaluate how therapeutic interventions in speech-language therapy impact the everyday lives of children and families by applying a theoretical framework. MAIN CONTRIBUTION: This discussion paper critically examines how the Bioecological Model of Development can be applied to the study of communicative participation outcomes following speech and language therapy. This model will be used in future analyses of communicative participation outcomes data in Ontario's PSLP. The strengths and challenges of this approach are reviewed.

Measure of processes of care: a review of 20 years of research.

AIM: This article reviews literature on findings from the Measure of Processes of Care (MPOC) to assess family-centred services. METHOD: Systematic searches for papers citing MPOC in both PubMed and Web of Science identified 107 articles. Fifty-five met the criterion for inclusion in this review in that they reported MPOC data. RESULTS: Over the past 20 years MPOC has been used in settings additional to the children's treatment centres for which it was designed; used in 11 countries and translated into 14 languages; and used to measure change in respondents' perceptions over time. MPOC findings have also informed our understanding of the provision of family-centred services. Overall, parents report that service providers do a good job of providing respectful, comprehensive services in partnership with families, but that there remain limitations in the provision of general information, an area for improvement. Finally, MPOC has been shown to correlate with various other measures related to the provision of family-centred services. INTERPRETATION: The MPOC 'family' of measures can be used to assess both families' and service providers' experiences and perceptions of the family-centredness of services received/provided. Opportunities abound for further research enquiries.

Age-related variables in childhood epilepsy: how do they relate to each other and to quality of life?

How do age of onset and duration of epilepsy correlate with each other and with patient-reported outcomes? To address this question, we explored whether age of onset, duration, and proportion of life with epilepsy are either similar or relatively independent variables that can be used as markers on how children experience the complexity of epilepsy and adjustment. Three hundred ninety-one Canadian and 266 Hong Kong youth with epilepsy completed the childhood epilepsy-specific quality of life (QOL) measure (CHEQOL-25). Each cohort was separately stratified by tertiles for age of onset, life proportion with epilepsy, and duration of epilepsy. Pearson's r was used for correlation analysis. The epilepsy age-related variables correlated strongly with each other among children with epilepsy onset ≤4 years (r = 0.53-0.66). The correlation between these variables was weaker with an onset ≥9 years (r =0.22-0.35). Correlation with QOL was clinically non-significant. These variables appear to measure the same phenomenon only in children with early epilepsy onset (<4 years) and explain little variance in QOL.

Criterion validity of the GMFM-66 item set and the GMFM-66 basal and ceiling approaches for estimating GMFM-66 scores.

AIM: The aim of this study was to compare the accuracy of two abbreviated approaches for estimating Gross Motor Function Measure 66 (GMFM-66) scores against the full GMFM-66 and to explore their strengths and limitations. METHOD: An existing dataset (n=224) comprising children aged 1 to 13 years (mean age 6y 11mo, SD 4y 6mo; 132 males, 92 females) with cerebral palsy (CP) of all Gross Motor Function Classification System (GMFCS) levels was used to compare the validity of the item set version (GMFM-66-IS) and the basal and ceiling version (GMFM-66-B&C) with the full GMFM-66 scores. Follow-up assessment at 1 year (n=109) allowed evaluation of change scores and accuracy at a single point in time. RESULTS: The cross-sectional agreement was excellent for both abbreviated measures (all intraclass correlation coefficients [ICCs] >0.98). When measuring change over time, both the GMFM-66-IS and the GMFM-66-B&C showed good agreement for children with bilateral CP (ICCs >0.9). However, the GMFM-66-IS assessed change over 1 year more accurately than the GMFM-66-B&C in children with unilateral CP (ICC=0.89 vs ICC=0.58; 95% confidence intervals do not overlap). INTERPRETATION: Both approaches for estimating GMFM-66 scores are accurate at a single point in time. If the primary goal of assessment is to measure change, the full GMFM-66 should still be regarded as the criterion standard. The GMFM-66-IS should be the preferred shortened measure for children with unilateral CP.

Patients and Families as Partners in Patient-Oriented Research: How Should They Be Compensated?

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.

Social participation of adolescents with cerebral palsy: trade-offs and choices.

This article reports on the findings of a qualitative study about the experiences and perceptions of adolescents with cerebral palsy (CP) in relation to social participation. A phenomenological approach was used to interview 10 adolescents with CP, 17 to 20 years of age, selected using purposeful sampling. An iterative process of data collection and analysis resulted in four themes about social participation. The themes of experience, barriers, and supports, and tradeoffs supported the current view of participation as a dynamic interaction between person and environment. The fourth theme of making choices described the unique challenges facing adolescents with CP in terms of deciding what was most important and meaningful to them now and in their future. Health care professionals can support adolescents as they develop the capacity to make their own decisions during the transition to adult living by ensuring that assessments and interventions address social participation.