Developing the FEEDS toolkit of parent-delivered interventions for eating, drinking and swallowing difficulties in young children with neurodisability: findings from a Delphi survey and stakeholder consultation workshops.

BACKGROUND: Young children with neurodisability commonly experience eating, drinking and swallowing difficulties (EDSD). Little is documented about which interventions and outcomes are most appropriate for such children. We aimed to seek consensus between parents of children with neurodisability and health professionals on the appropriate interventions and outcomes to inform future clinical developments and research studies. METHODS: Two populations were sampled: parents of children aged up to 12 years with neurodisability who experienced EDSD; health professionals working with children and young people (aged 0-18 years) with neurodisability with experience of EDSD. Participants had taken part in a previous national survey and were invited to take part in a Delphi survey and/or consultation workshops. Two rounds of this Delphi survey sought agreement on the appropriate interventions and outcomes for use with children with neurodisability and EDSD. Two stakeholder consultation workshops were iterative, with the findings of the first discussed at the second, and conclusions reached. RESULTS: A total of 105 parents and 105 health professionals took part. Parents and health professionals viewed 19 interventions and 10 outcomes as essential. Interventions related to improvement in the physical aspects of a child's EDSD, behavioural changes of the child or parent, and changes in the child or family's well-being. Both parents and health professionals supported a 'toolkit' of interventions that they could use together in shared decision making to prioritise and implement timely interventions appropriate to the child. CONCLUSIONS: This study identified interventions viewed as essential to consider for improving EDSD in children with neurodisability. It also identified several key outcomes that are valued by parents and health professionals. The Focus on Early Eating, Drinking and Swallowing (FEEDS) Toolkit of interventions to improve EDSD in children with neurodisability has been developed and now requires evaluation regarding its use and effectiveness.

Parent-delivered interventions used at home to improve eating, drinking and swallowing in children with neurodisability: the FEEDS mixed-methods study.

BACKGROUND: Eating, drinking and swallowing difficulties are common in young children with neurodisability. These difficulties may lead to inadequate calorie intake, which affects a child's nutrition, growth and general physical health. OBJECTIVE: To examine which interventions are available that can be delivered at home by parents to improve eating, drinking and swallowing in young children with neurodisability and are suitable for investigation in pragmatic trials. DESIGN: This was a mixed-methods study that included focus groups, surveys, an update of published systematic reviews of interventions, a systematic review of measurement properties of existing tools, evidence mapping, evidence synthesis, a Delphi survey and stakeholder workshops. SETTING: The study was carried out in NHS hospitals, community services, family homes and schools. PARTICIPANTS: Parents of children who had neurodisability and eating, drinking and swallowing difficulties. Professionals from health and education. Young people with eating, drinking and swallowing difficulties or young people who had previously experienced eating, drinking and swallowing difficulties. DATA SOURCES: Literature reviews; national surveys of parents and professionals; focus groups with parents, young people and professionals; and stakeholder consultation workshops. REVIEW METHODS: An update of published systematic reviews of interventions (searched July-August 2017), a mapping review (searched October 2017) and a systematic review of measurement properties using COnsensus-based Standards for the Selection of health status Measurement INstruments (COSMIN) methodology (searched May 2018). RESULTS: Significant limitations of the available research evidence regarding interventions and tools to measure outcomes were identified. A total of 947 people participated: 400 parents, 475 health professionals, 62 education professionals and 10 young people. The survey showed the wide range of interventions recommended by NHS health professionals, with parents and professionals reporting variability in the provision of these interventions. Parents and professionals considered 19 interventions as relevant because they modified eating, drinking and swallowing difficulties. Parents and professionals considered 10 outcomes as important to measure (including Nutrition, Growth and Health/safety); young people agreed that these were important outcomes. Stakeholder consultation workshops identified that project conclusions and recommendations made sense, were meaningful and were valued by parents and professionals. Parents and health professionals were positive about a proposed Focus on Early Eating, Drinking and Swallowing (FEEDS) toolkit of interventions that, through shared decision-making, could be recommended by health professionals and delivered by families. LIMITATIONS: The national surveys included large numbers of parents and professionals but, as expected, these were not representative of the UK population of parents of children with eating, drinking and swallowing difficulties. Owing to the limitations of research evidence, pragmatic decisions were made about interventions that might be included in future research and outcomes that might be measured. For instance, the reviews of research found only weak or poor evidence to support the effectiveness of interventions. The review of outcome measures found only limited low-level evidence about their psychometric properties. CONCLUSIONS: Opportunities and challenges for conducting clinical trials of the effectiveness of the FEEDS toolkit of interventions are described. Parents and professionals thought that implementation of the toolkit as part of usual NHS practice was appropriate. However, this would first require the toolkit to be operationalised through development as a complex intervention, taking account of constituent interventions, delivery strategies, implementation and manualisation. Subsequently, an evaluation of its clinical effectiveness and cost-effectiveness could be undertaken using appropriate research methods. FUTURE WORK: Initial steps include FEEDS toolkit development and evaluation of its use in clinical practice, and identification of the most robust methods to measure valued outcomes, such as Nutrition and Growth. TRIAL REGISTRATION: Current Controlled Trials ISRCTN10454425. FUNDING: This project was funded by the National Institute for Health Research (NIHR) Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 25, No. 22. See the NIHR Journals Library website for further project information.

Some young children with disabilities, such as those with cerebral palsy and autism spectrum disorder, have difficulties with eating, drinking and swallowing. Many strategies are employed to help with these difficulties (e.g. 'Positioning'), but we need to know more about which ones work best. We wanted to find out about the strategies parents use at home to help their children with eating, drinking and swallowing difficulties. We wanted to understand what is already known about the strategies that are available now and if children are getting the right help. We wanted to find out if doing more research would tell us which interventions work. We looked at what has been written about the ways in which parents help their child to eat, drink and swallow. We discussed this information with parents and health professionals. We developed a survey to ask what parent-delivered strategies are recommended by NHS professionals, which strategies parents use and how we would know if things had improved. We discussed the findings with parents and professionals. We then used a different type of survey. Parents and professionals were asked which strategies were most important and what they would most like to improve. We then held workshops to hear parents' and professionals' views on what we had found and to agree on how we would organise future research. We discussed some of the findings with young people. Research about these strategies and how to measure improvements in eating and drinking is of poor quality. A wide variety of strategies are used: 19 strategies were thought to be the most useful by parents and professionals. Parents and professionals agreed on 10 areas that they would most like to improve. Both groups thought that it was a good idea to have a 'toolkit' of strategies so that they could choose the right strategy at the right time.

Disparity of child/parent-reported quality of life in cerebral palsy persists into adolescence.

AIM: To examine the evolution of child-parent discrepancy in reporting quality of life (QoL) between childhood and adolescence in children with cerebral palsy (CP) and to investigate potential factors associated with such a discrepancy. METHOD: We used data from the SPARCLE (Study of PARticipation of Children with CP Living in Europe) study, a population-based cohort study of children with CP, aged 8 to 12 years at baseline (in 2004-2005), in nine European centres, who were followed up at the age of 13 to 17 years. The KIDSCREEN-52 Quality of Life measure was used at baseline and follow-up; 354 child-parent dyads out of 500 eligible dyads were followed up (201 males, 153 females). We used intraclass correlation coefficients (ICCs) to examine agreement between parent proxy-reports and self-reported QoL. We used linear regression to examine factors associated with child-parent discrepancy in QoL reporting. RESULTS: Agreement was low to moderate (ICC=0.16-0.48) in childhood and in adolescence across all QoL domains. In four domains (moods and emotions, self-perception, relationship with parents and home life, and social support and peers), the extent of the discrepancy increased significantly between childhood and adolescence. Parenting stress, child pain, and child behaviour problems influenced parent proxy-reports during both childhood and adolescence. INTERPRETATION: The points of view of the child and their parents should be treated as complementary to obtain better knowledge regarding the QoL of children and adolescents with CP.

Did previous involvement in research affect recruitment of young people with cerebral palsy to a longitudinal study of transitional health care?

OBJECTIVE: To assess whether being contacted about or participating in previous research and method of approaching potential participants affected recruitment to a transition study from child to adult healthcare services of young people with cerebral palsy (CP). DESIGN AND METHODS: Young people with CP aged 14-18 years without severe intellectual impairment were identified from regional registers of CP in Northern Ireland and the North of England. χ2 and Mann-Whitney U tests were used to assess differences in CP and sociodemographic characteristics between those recruited and those who refused. Logistic regression was used to assess contact about and recruitment to previous research and method of approach as predictors of recruitment, controlling for demographic and CP characteristics. RESULTS: Of the 410 young people who were approached; 162 did not respond and of the 248 who responded, 96 (23%) were recruited. There were significant differences between those recruited and those who refused in age and number of previous studies they had participated in. Those who were older or who had previously been approached about research were more likely to be recruited to our study. However, those who had been recruited to previous studies were more likely to refuse to join our study. CONCLUSIONS: The method of approach to potential participants did not affect recruitment. Older adolescents and those who had been approached about previous research were more likely to take part in our study, although there was evidence of research fatigue because if they had actually been recruited to the previous studies they were less likely to join our study. Recruitment of adolescents to studies remains challenging.

Facilitating transition of young people with long-term health conditions from children's to adults' healthcare services - implications of a 5-year research programme.

BACKGROUND: During transition from children's to adults' healthcare, young adults with long-term conditions may show delays in psychosocial development compared to their peers without long-term conditions, and deterioration of their conditions' medical control. METHODS: This paper integrates the findings, already published in 10 separate papers, of a 5-year transition research programme. IMPLICATIONS: There is an important role for funders (commissioners) of adults' services to fund transitional healthcare, in addition to funders of children's services who currently take responsibility.It is important that healthcare provider organisations adopt an organisation-wide approach to implementation to ensure that good practice is adopted in children's and adults' services, not just adopted by enthusiasts in some specialties. This includes provision of 'developmentally appropriate healthcare' which recognises the changing biopsychosocial developmental needs of young people.Three features of transitional healthcare were associated with improved outcomes: appropriate parent involvement, promotion of young people's confidence in managing their health and meeting the adult team before transfer. These should be maintained or introduced as a priority.Child and adult healthcare providers should routinely explore with a young person how they approach transition and personalise their clinical approach thereafter.These implications are relevant for a range of stakeholders, including funders of transitional healthcare, organisations providing transitional healthcare and clinical practitioners.

Standing frames for children with cerebral palsy: a mixed-methods feasibility study.

BACKGROUND: Standing frames are recommended as part of postural management for young people with cerebral palsy (CP) Gross Motor Function Classification System (GMFCS) level IV or V. They may have a variety of benefits, including improving bone mineral density, gastrointestinal function and social participation. The NHS needs to know if these benefits are real, given the cost implications of use and the reported negative effects (e.g. pain). The lack of evidence for the clinical effectiveness of standing frames demonstrates the need for evaluative research. OBJECTIVE(S): The aim of the study was to explore the acceptability of a future trial to determine the clinical effectiveness of standing frames. DESIGN: A sequential mixed-methods design was used. The findings of each stage informed the next stage. We conducted surveys, focus groups and in-depth interviews. PARTICIPANTS: Professionals who work with young people who use standing frames and parents who have a child who uses a standing frame took part in a survey of current standing frame practice (n = 551), a series of focus groups (seven focus groups, 49 participants in total) and a survey of research trial acceptability and feasibility (n = 585). Twelve young people who use a standing frame were interviewed. RESULTS: Standing frames were widely used as part of postural management for young people with CP both in school and at home but more frequently in school, and particularly by young people in primary school. Achieving the prescribed use was not always possible owing to resources, environment and family factors. Participation and activity engagement were important to young people. The majority of participants believed that standing frames research is necessary. Some reported concern that stopping standing frame use for a trial would cause irreversible damage. The maximum amount of time most health professionals and parents would agree to suspend standing frame use would be 12 weeks. LIMITATIONS: Owing to the nature of recruitment, we could not calculate response rates or determine non-response bias. Therefore, participants may not be representative of all standing frame users. CONCLUSIONS: Although parents and professionals who engaged in the qualitative aspect of this research and stakeholders who took part in the design workshops appreciated the lack of clinical evidence, our surveys, qualitative information and PPI demonstrated that most people had strong beliefs regarding the clinical effectiveness of standing frames. However, with key stakeholder engagement and careful planning, a trial would be acceptable. FUTURE WORK: We recommend a carefully planned trial that includes a pilot phase. The trial should evaluate the following question: 'does using a standing frame in school improve patient-reported outcomes of participation (primary outcome), quality of life, subjective well-being, body function and body structure (secondary outcomes) in young children (aged 4-11 years) with CP GMFCS III-V?'. FUNDING: The National Institute for Health Research Health Technology Assessment programme.

Falling through the gaps: exploring the role of integrated commissioning in improving transition from children's to adults' services for young people with long-term health conditions in England.

Objectives To explore the role of integrated commissioning in improving the transition of young people with long-term conditions from child to adult services. We aimed to identify organizational and policy gaps around transition services and provide recommendations for integrated commissioning practice. Methods Semi-structured in-depth interviews were conducted with two groups of participants: (1) twenty-four stakeholders involved in the commissioning and provision of transition services for young people with long-term conditions in two regions in England; (2) five professionals with national roles in relation to planning for transition. Transcripts were interrogated using thematic analysis. Results There is little evidence of integrated commissioning for transitional care for young people with long-term conditions. Commissioners perceive there to be a lack of national and local policy to guide integrated commissioning for transitional care; and limited resources for transition. Furthermore, commissioning organizations responsible for transition have different cultures, funding arrangements and related practices which make inter- and intra-agency co-ordination and cross-boundary continuity of care difficult to achieve. Conclusions Integrated commissioning may be an effective way to achieve successful transitional care for young people with long-term health conditions. However, this innovative relational approach to commissioning requires a national steer together with recognition of common values and joint ownership between relevant stakeholders.

Drooling Reduction Intervention randomised trial (DRI): comparing the efficacy and acceptability of hyoscine patches and glycopyrronium liquid on drooling in children with neurodisability.

OBJECTIVE: Investigate whether hyoscine patch or glycopyrronium liquid is more effective and acceptable to treat drooling in children with neurodisability. DESIGN: Multicentre, single-blind, randomised controlled trial. SETTING: Recruitment through neurodisability teams; treatment by parents. PARTICIPANTS: Ninety children with neurodisability who had never received medication for drooling (55 boys, 35 girls; median age 4 years). EXCLUSION CRITERIA: medication contraindicated; in a trial that could affect drooling or management. INTERVENTION: Children were randomised to receive a hyoscine skin patch or glycopyrronium liquid. Dose was increased over 4 weeks to achieve optimum symptom control with minimal side-effects; steady dose then continued to 12 weeks. PRIMARY AND SECONDARY OUTCOMES: Primary outcome: Drooling Impact Scale (DIS) score at week-4. SECONDARY OUTCOMES: change in DIS scores over 12 weeks, Drooling Severity and Frequency Scale and Treatment Satisfaction Questionnaire for Medication; adverse events; children's perception about treatment. RESULTS: Both medications yielded clinically and statistically significant reductions in mean DIS at week-4 (25.0 (SD 22.2) for hyoscine and 26.6 (SD 16) for glycopyrronium). There was no significant difference in change in DIS scores between treatment groups. By week-12, 26/47 (55%) children starting treatment were receiving hyoscine compared with 31/38 (82%) on glycopyrronium. There was a 42% increased chance of being on treatment at week-12 for children randomised to glycopyrronium relative to hyoscine (1.42, 95% CI 1.04 to 1.95). CONCLUSIONS: Hyoscine and glycopyrronium are clinically effective in treating drooling in children with neurodisability. Hyoscine produced more problematic side effects leading to a greater chance of treatment cessation. TRIAL REGISTRATION NUMBERS: ISRCTN 75287237; EUDRACT: 2013-000863-94; Medicines and Healthcare Products Regulatory Agency: 17136/0264/001-0003.

What constitutes successful commissioning of transition from children's to adults' services for young people with long-term conditions and what are the challenges? An interview study.

OBJECTIVE: We explored what constitutes successful commissioning for transition and what challenges are associated with this. We aimed: (1) to identify explicit and implicit organisational structures, processes and relationships that drive commissioning around transition; (2) to identify challenges faced by commissioners; and (3) to develop a conceptual model. DESIGN: A qualitative interview study. SETTING: Commissioning and provider organisations across primary and secondary care and third sector in England, UK. PARTICIPANTS: Representatives (n=14) from clinical commissioning groups, health and well-being boards and local authorities that commission national health services (NHS) for transition from children's to adults' services in England; NHS directors, general practitioners and senior clinicians (n=9); and frontline NHS and third sector providers (n=6). RESULTS: Both commissioners and providers thought successful transition is personalised, coordinated and collaborative with a focus on broad life outcomes and actualised through building pathways and universal services. A multitude of challenges were described, including inconsistent national guidance, fragmented resources, incompatible local processes, lack of clear outcomes and professional roles and relationships. No single specific process of commissioning for transition emerged-instead complex, multi-layered, interactive processes were described. CONCLUSIONS: The findings indicate a need to consider more explicitly the impact of national policies and funding streams on commissioning for transition. Commissioners need to require care pathways that enable integrated provision for this population and seek ways to ensure that generalist community providers engage with children with long-term conditions from early on. Future research is needed to identify a core set of specific, meaningful transition outcomes that can be commissioned, measured and monitored.

How available to European children and young people with cerebral palsy are features of their environment that they need?

BACKGROUND: The UN Convention on the Rights of Persons with Disabilities requires accessibility to the physical and social environments. However, individuals with cerebral palsy (CP) have many difficulties in accessing the environment they need for functional independence and social inclusion. AIMS: To examine the availability of environmental features which children with CP need for optimal participation, and whether availability changed for them between ages 8-12 and 13-17 years. METHODS: The sample is the 594 children with CP, born 31/07/1991-01/04/1997, who took part in the SPARCLE study at age 8-12 (SPARCLE 1) and again at 13-17 years (SPARCLE 2). Participants were randomly sampled from population registers of children with CP in eight European regions; one further region recruited from multiple sources. Data about environment were captured with the European Child Environment Questionnaire (60 items). Differences in availability of environmental features between childhood and adolescence were assessed using McNemar's test; differences between regions were assessed by ranking regions. Differences in availability between regions were assessed by ranking regions. RESULTS: For seven environmental features significantly (p<0.01) fewer individuals needed the feature in SPARCLE 2 than in SPARCLE 1, whilst for two features more individuals needed the feature. Nine features in SPARCLE 1 and six features in SPARCLE 2 were available to less than half the participants who needed them. Eight features showed significantly (p<0.01) higher availability in SPARCLE 2 than in SPARCLE 1 (enlarged rooms, adapted toilet, modified kitchen and hoists at home, adapted toilets and lifts at school, an adequate vehicle, grants for home modifications) while none showed significantly lower availability. The relative rankings of the better and less good regions persisted from the age 8-12year age group to the 13-17year age group. CONCLUSIONS: Needed environmental features are unavailable to many children at ages 8-12 and 13-17 years. This lack of availability is more pronounced in some regions than others, which probably results from their policy, legislative and statutory frameworks.

Predictors of parent-reported quality of life of adolescents with cerebral palsy: A longitudinal study.

AIM: Parent-reporting is needed to examine Quality of Life (QoL) of children with cerebral palsy (CP) across all severities. This study examines whether QoL changes between childhood and adolescence, and what predicts adolescent QoL. METHOD: SPARCLE is a European cohort study of children with CP, randomly sampled from population databases. Of 818 8-12-year-olds joining the study, 594 (73%) were revisited as 13-17-year-olds. The subject of this report is the 551 (316 boys, 235 girls) where the same parent reported QoL on both occasions using KIDSCREEN-52 (transformed Rasch scale, mean 50, SD 10 per domain). Associations were assessed using linear regression. RESULTS: Between childhood and adolescence, average QoL reduced in six domains (1.3-3.8 points, p<0.01) and was stable in three (Physical wellbeing, Autonomy, Social acceptance). Socio-demographic factors had little predictive value. Childhood QoL was a strong predictor of all domains of adolescent QoL. Severe impairments of motor function, IQ or communication predicted higher adolescent QoL on some domains; except that severe motor impairment predicted lower adolescent QoL on the Autonomy domain. More psychological problems and higher parenting stress in childhood and their worsening by adolescence predicted lower QoL in five and eight domains respectively; contemporaneous pain in seven domains. The final model explained 30%-40% of variance in QoL, depending on domain. INTERPRETATION: In general, impairment severity and socio-demographic factors were not predictors of lower adolescent QoL. However, pain, psychological problems and parenting stress were predictors of lower adolescent QoL in most domains. These are modifiable factors and addressing them may improve adolescent QoL.

Views of Young People With Chronic Conditions on Transition From Pediatric to Adult Health Services.

PURPOSE: This study sought to identify and describe the views of young people with chronic conditions about the transition from pediatric to adult services. METHODS: Q methodology was used to identify young people's views on transition. A set of 39 statements about transition was developed from an existing literature review and refined in consultation with local groups of young people. Statements were printed onto cards and a purposive sample of 44 young people with chronic health conditions was recruited, 41 remaining in the study. The young people were asked to sort the statement cards onto a Q-sort grid, according to their opinions from "strongly disagree" to "strongly agree." Factor analysis was used to identify shared points of view (patterns of similarity between individual's Q-sorts). RESULTS: Four distinct views on transition were identified from young people: (1) "a laid-back view of transition;" (2) "anxiety about transition;" (3) "wanting independence and autonomy during transition;" and (4) "valuing social interaction with family, peers, and professionals to assist transition." CONCLUSIONS: Successful transition is likely to be influenced by how young people view the process. Discussing and understanding young people's views and preferences about transition should help clinicians and young people develop personalized planning for transition as a whole, and more specifically the point of transfer, leading to effective and efficient engagement with adult care.

Cerebral palsy.

Cerebral palsy is the most common cause of childhood-onset, lifelong physical disability in most countries, affecting about 1 in 500 neonates with an estimated prevalence of 17 million people worldwide. Cerebral palsy is not a disease entity in the traditional sense but a clinical description of children who share features of a non-progressive brain injury or lesion acquired during the antenatal, perinatal or early postnatal period. The clinical manifestations of cerebral palsy vary greatly in the type of movement disorder, the degree of functional ability and limitation and the affected parts of the body. There is currently no cure, but progress is being made in both the prevention and the amelioration of the brain injury. For example, administration of magnesium sulfate during premature labour and cooling of high-risk infants can reduce the rate and severity of cerebral palsy. Although the disorder affects individuals throughout their lifetime, most cerebral palsy research efforts and management strategies currently focus on the needs of children. Clinical management of children with cerebral palsy is directed towards maximizing function and participation in activities and minimizing the effects of the factors that can make the condition worse, such as epilepsy, feeding challenges, hip dislocation and scoliosis. These management strategies include enhancing neurological function during early development; managing medical co-morbidities, weakness and hypertonia; using rehabilitation technologies to enhance motor function; and preventing secondary musculoskeletal problems. Meeting the needs of people with cerebral palsy in resource-poor settings is particularly challenging.

Classification systems of communication for use in epidemiological surveillance of children with cerebral palsy.

AIM: Children with cerebral palsy (CP) often experience communication difficulties. We aimed to identify a classification system for communication of children with CP suitable for epidemiological surveillance. METHOD: Systems to classify the communication of children with CP were identified. The Communication Function Classification System (CFCS), Functional Communication Classification System (FCCS), and Viking Speech Scale (VSS) were chosen for further investigation and translated. They were administered to 155 children aged 4 to 13 years with CP (across all motor severity levels) from eight European countries. Children's parents/carers, speech therapists, and other health professionals applied the systems through direct observation. Other professionals applied them from case notes only. The systems were assessed for agreement, stability, ease, and feasibility of application. RESULTS: Test-retest stability was moderate-to-high for VSS (k=0.66-0.88), CFCS (k=uncomputed-0.91), and FCCS (k=0.52-0.91). Overall interrater agreement was fair to very good for every classification system. VSS achieved the best agreement between parents/carers and speech therapists. VSS was considered the easiest instrument to apply. INTERPRETATION: Because of its ease of use by a range of healthcare professionals, the VSS should be considered for CP registers which intend to survey speech intelligibility. For a wider assessment of communication, the CFCS or FCC should be considered.

Item response theory and structural equation modelling for ordinal data: Describing the relationship between KIDSCREEN and Life-H.

Both item response theory and structural equation models are useful in the analysis of ordered categorical responses from health assessment questionnaires. We highlight the advantages and disadvantages of the item response theory and structural equation modelling approaches to modelling ordinal data, from within a community health setting. Using data from the SPARCLE project focussing on children with cerebral palsy, this paper investigates the relationship between two ordinal rating scales, the KIDSCREEN, which measures quality-of-life, and Life-H, which measures participation. Practical issues relating to fitting models, such as non-positive definite observed or fitted correlation matrices, and approaches to assessing model fit are discussed. item response theory models allow properties such as the conditional independence of particular domains of a measurement instrument to be assessed. When, as with the SPARCLE data, the latent traits are multidimensional, structural equation models generally provide a much more convenient modelling framework.

Characteristics of young people with long term conditions close to transfer to adult health services.

BACKGROUND: For many young people with long term conditions (LTC), transferring from paediatric to adult health services can be difficult and outcomes are often reported to be poor. We report the characteristics and representativeness of three groups of young people with LTCs as they approach transfer to adult services: those with autism spectrum disorder with additional mental health problems (ASD); cerebral palsy (CP); or diabetes. METHODS: Young people aged 14 years-18 years 11 months with ASD, or those with diabetes were identified from children's services and those with CP from population databases. Questionnaires, completed by the young person and a parent, included the 'Mind the Gap' Scale, the Rotterdam Transition Profile, and the Warwick and Edinburgh Mental Wellbeing Scale. RESULTS: Three hundred seventy four young people joined the study; 118 with ASD, 106 with CP, and 150 with diabetes. Participants had a significant (p < 0.001) but not substantial difference in socio-economic status (less deprived) compared to those who declined to take part or did not respond. Condition-specific severity of participants was similar to that of population data. Satisfaction with services was good as the 'gap' scores (the difference between their ideal and current care) reported by parents and young people were small. Parents' satisfaction was significantly lower than their children's (p < 0.001). On every domain of the Rotterdam Transition Profile, except for education and employment, significant differences were found between the three groups. A larger proportion of young people with diabetes were in a more independent phase of participation than those with ASD or CP. The wellbeing scores of those with diabetes (median = 53, IQR: 47-58) and CP (median = 53, IQR: 48-60) were similar, and significantly higher than for those with ASD (median = 47, IQR: 41-52; p < 0.001). CONCLUSIONS: Having established that our sample of young people with one of three LTCs recruited close to transfer to adult services was representative, we have described aspects of their satisfaction with services, participation and wellbeing, noting similarities and differences by LTC. This information about levels of current functioning is important for subsequent evaluation of the impact of service features on the health and wellbeing of young people with LTCs following transfer from child services to adult services.

Predictors of participation of adolescents with cerebral palsy: A European multi-centre longitudinal study.

We investigated whether childhood factors that are amenable to intervention (parenting stress, child psychological problems and pain) predicted participation in daily activities and social roles of adolescents with cerebral palsy (CP). We randomly selected 1174 children aged 8-12 years from eight population-based registers of children with CP in six European countries; 743 (63%) agreed to participate. One further region recruited 75 children from multiple sources. These 818 children were visited at home at age 8-12 years, 594 (73%) agreed to follow-up at age 13-17 years. We used the following measures: parent reported stress (Parenting Stress Index Short Form), their child's psychological difficulties (Strength and Difficulties Questionnaire) and frequency and severity of pain; either child or parent reported the child's participation (LIFE Habits questionnaire). We fitted a structural equation model to each of the participation domains, regressing participation in childhood and adolescence on parenting stress, child psychological problems and pain, and regressing adolescent factors on the corresponding childhood factors; models were adjusted for impairment, region, age and gender. Pain in childhood predicted restricted adolescent participation in all domains except Mealtimes and Communication (standardized total indirect effects ß -0.05 to -0.18, 0.01

Self-reported quality of life of adolescents with cerebral palsy: a cross-sectional and longitudinal analysis.

BACKGROUND: Children with cerebral palsy who can self-report have similar quality of life (QoL) to their able-bodied peers. Is this similarity also found in adolescence? We examined how self-reported QoL of adolescents with cerebral palsy varies with impairment and compares with the general population, and how factors in childhood predict adolescent QoL. METHODS: We report QoL outcomes in a longitudinal follow-up and cross-sectional analysis of individuals included in the SPARCLE1 (childhood) and SPARCLE2 (adolescent) studies. In 2004 (SPARCLE1), a cohort of 818 children aged 8-12 years were randomly selected from population-based cerebral palsy registers in nine European regions. We gathered data from 500 participants about QoL with KIDSCREEN (ten domains); frequency of pain; child psychological problems (Strengths and Difficulties Questionnaire); and parenting stress (Parenting Stress Index). At follow-up in 2009 (SPARCLE2), 355 (71%) adolescents aged 13-17 years remained in the study and self-reported QoL (longitudinal sample). 76 additional adolescents self-reported QoL in 2009, providing data for 431 adolescents in the cross-sectional sample. Researchers gathered data at home visits. We compared QoL against matched controls in the general population. We used multivariable regression to relate QoL of adolescents with cerebral palsy to impairments (cross-sectional analysis) and to childhood QoL, pain, psychological problems, and parenting stress (longitudinal analysis). FINDINGS: Severity of impairment was significantly associated (p<0·01) with reduced adolescent QoL on only three domains (Moods and emotions, Autonomy, and Social support and peers); average differences in QoL between the least and most able groups were generally less than 0·5 SD. Adolescents with cerebral palsy had significantly lower QoL than did those in the general population in only one domain (Social support and peers; mean difference -2·7 [0·25 SD], 95% CI -4·3 to -1·4). Pain in childhood or adolescence was strongly associated with low adolescent QoL on eight domains. Childhood QoL was a consistent predictor of adolescent QoL. Child psychological problems and parenting stress in childhood or their worsening between childhood and adolescence predicted only small reductions in adolescent QoL. INTERPRETATION: Individual and societal attitudes should be affected by the similarity of the QoL of adolescents with and without cerebral palsy. Adolescents with cerebral palsy need particular help to maintain and develop peer relationships. Interventions in childhood to alleviate psychological difficulties, parenting stress, and especially pain, are justified for their intrinsic value and for their longer term effect on adolescent QoL. FUNDING: SPARCLE1 was funded by the European Union Research Framework 5 Program (grant number QLG5-CT-2002-00636), the German Ministry of Health GRR-58640-2/14, and the German Foundation for the Disabled Child. SPARCLE2 was funded by: Wellcome Trust WT086315 A1A (UK and Ireland); Medical Faculty of the University of Lübeck E40-2009 and E26-2010 (Germany); CNSA, INSERM, MiRe-DREES, and IRESP (France); Ludvig and Sara Elsass Foundation, The Spastics Society and Vanforefonden (Denmark); Cooperativa Sociale "Gli Anni in Tasca" and Fondazione Carivit, Viterbo (Italy); Göteborg University-Riksforbundet for Rorelsehindrade Barn och Ungdomar and the Folke Bernadotte Foundation (Sweden).