Assessing the Adequacy of the Physical, Social, and Attitudinal Environment to the Specific Needs of Young Adults With Cerebral Palsy: The European Adult Environment Questionnaire.

OBJECTIVES: To present the development of the European Adult Environment Questionnaire (EAEQ), to assess to what extent it covers the International Classification of Functioning, Disability and Health (ICF), and to describe the adequacy of the physical, social, and attitudinal environment to the specific needs of young adults with cerebral palsy (CP). DESIGN: Cross-sectional. SETTING: Administrative regions in France, Germany, Italy, Portugal, and Sweden. PARTICIPANTS: Young adults with CP (N=357), with varying severity profiles, aged 19-28 years at time of interview (2018-20). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURE(S): Physical, social, and attitudinal environment unmet needs. RESULTS: Relevant environmental factors (EFs) for young adults with CP were identified during focus groups in England and Portugal. EFs were mapped to the ICF environmental classification and the EAEQ analytical structure resulted from this linking procedure. It comprised 61 items, linked to 31 ICF environmental classification categories, and covered 4 of its 5 chapters. Content validity assessed with the bandwidth index (percentage coverage of ICF Core Sets for adults with CP) was satisfactory (79.3%). A descriptive analysis was carried out. Participants had a mean age of 24 years, 56% were men, 38% had severely limited mobility. Less than 16% reported unmet needs for EFs relating to home, college/work/day placement, and communication in the Products and technology chapter. Unmet needs were higher (>20%) for the other items in the Public use and Land development categories. Social support, attitudes, and understanding of relatives were often adequate to the participants' needs. The proportion of unmet needs varied by sex (women were more often concerned) and raised with increasing gross motor impairment. CONCLUSION: The EAEQ describes in detail the adequacy of the environment to the specific needs of young adults with CP. Its ICF-based structure opens up possibilities for use in a universal conceptual framework.

A systematic review and meta-analysis of school and cognitive function domains of health-related quality of life measures for children and young adults with congenital heart disease.

BACKGROUND: Research on cognitive and school functioning domains of health-related quality of life (HRQOL) for children and adolescents with congenital heart disease (CHD) presents inconsistencies. OBJECTIVES: To summarize and synthesize data on school and cognitive function domains of HRQOL for children and young people (CYP) with CHD. METHODS: Five electronic databases MEDLINE, Scopus, PsycINFO, EMBASE, ERI, and citations were systematically searched. We included original-research articles reporting the cognitive and school function domains of HRQOL for children and young people with CHD (child and parent reports included). Both fixed and random-effects meta-analyses were performed to estimate pooled mean test scores for cognitive and school function. A total of 34 studies met our inclusion criteria and were synthesized narratively, 17 studies were included in formal meta-analyses. RESULTS: Self-reported cognitive function was lower for children and young people with CHD than healthy controls (SMD -0.28 (-0.42, -0.15)). Parental reports demonstrated similar results to self-reports (SMD -0.54 (-0.91, -0.18)). School function was lower in children and young people with CHD compared with healthy controls in self-reported (SMD -0.30 (-0.48, -0.13)) and parent reported HRQOL (SMD -0.49 (0.64, -0.36)). Self-reported school function domain scores were lower for young (<8 years) (SMD -0.65 (-1.32, 0.03)) and older children (8-18 years) (SMD -0.25 (-0.47, -0.03)) with CHD than their peers. Similarly, parents reported lower school function domain scores for young (<8 years) (SMD -0.68 (-1.29, -0.07)) and older (8-18 years) (SMD -0.46 (-068, -0.25)) children with CHD than typically developing peers. CONCLUSION: Children born with CHD may experience lower cognitive and school function HRQOL scores than healthy controls (self and proxy-report). This is consistent with a subgroup meta-analysis of young (<8 years) and older (8 years old or more) children with CHD reporting lower school function scores compared to controls.

Maternal age and the prevalence of congenital heart defects in Europe, 1995-2015: A register-based study.

BACKGROUND: Evidence on the direction and strength of association between maternal age and the prevalence of congenital heart defects (CHD) in different age group categories is conflicting. Some studies have illustrated different trends with an increase in prevalence in younger and older age groups while other studies have reported a linear relationship. Given the increase in maternal age over recent years, it is important to study the CHD prevalence by maternal age. OBJECTIVES: To examine the association between maternal age and the prevalence of CHD in Europe between 1995 and 2015 using population-based data from 24 registries belonging to the European Surveillance of Congenital Anomalies (EUROCAT) network. METHODS: Associations over time of all nonsyndromic CHD according to maternal age category and for three CHD severity groupings (severity group I: very severe; severity group II: severe; severity group III: less severe) were examined using Bayesian multilevel Poisson regression modeling. Further subgroup analyses were undertaken within four maternal age-bands: ≤24, 25-29, 30-34 and 35-44 years. Descriptive summaries are also presented. RESULTS: There were 51,608 nonsyndromic CHD cases in Europe over the 20-year study period. Total prevalence for all CHD combined was increased for younger mothers (≤24 years) and for mothers 35-44 years of age when compared with mothers aged 25-29 years (reference group) (IRR: 1.05, 95% CI: 1.02, 1.07). The total prevalence was increased for severity group I (very severe) only for younger mothers compared to those aged 25-29 years (IRR: 1.14, 95% CI: 1.04, 1.23). We found an increased prevalence of the following CHD subtypes: double outlet right ventricle (IRR:1.33, 95% CI: 1.09, 1.60), hypoplastic left heart syndrome (IRR: 1.18, 95% CI: 1.05, 1.32), hypoplastic right heart syndrome (IRR: 1.41, 95% CI: 1.05, 1.84), atrioventricular septal defect (IRR: 1.15, 95% CI: 1.01, 1.32), coarctation of aorta (IRR: 1.15, 95% CI: 1.03, 1.28) and atrial septal defect (IRR: 1.08, 95% CI: 1.02, 1.13). For older mothers (35-44 years) compared to the reference category, we observed an increased risk in the prevalence for severity group II (IRR: 1.09, 95% CI: 1.03, 1.14), severity group III (IRR: 1.05, 95% CI: 1.01, 1.08) and an increased prevalence of the CHD subtypes: Pulmonary valve stenosis (IRR: 1.22, 95% CI: 1.09, 1.34), ASD (IRR: 1.07, 95% CI: 1.02, 1.13), CoA (IRR: 1.18, 95% CI: 1.06, 1.32) and Tetralogy of Fallot (IRR: 1.14, 95% CI: 1.01, 1.28). Finally, for all age categories compared to the reference category, different associations of ASD and an increased prevalence of CoA was also observed. CONCLUSIONS: Based on data for cases of CHD from 24 European population-based registries, evidence of a positive association between maternal age and the total prevalence of CHD for younger (≤24 years old) and older (35-44 years old) mothers was observed. The results suggest that young maternal age (≤24 years old) is a factor associated with severe CHD phenotypes while a positive association between advanced maternal age (35-44 years old) and mild CHD phenotypes was observed.

Impacts of health care service changes implemented due to COVID-19 on children and young people with long-term disability: A mapping review.

AIM: To identify the research on childhood disability service adaptations and their impact on children and young people with long-term disability during the COVID-19 pandemic. METHOD: A mapping review was undertaken. We searched the World Health Organization Global COVID-19 database using the search terms 'children', 'chronic/disabling conditions', and 'services/therapies'. Eligible papers reported service changes for children (0-19 years) with long-term disability in any geographical or clinical setting between 1st January 2020 and 26th January 2022. Papers were charted across the effective practice and organization of care taxonomy of health system interventions and were narratively synthesized; an interactive map was produced. RESULTS: Reduction of face-to-face care and usual provision had a huge impact on children and families. Adoption of telehealth provided continuity for the care and management of some conditions. There was limited evidence of changes to mental health services, transitions of care, social care, or child-reported satisfaction or acceptability of service changes. INTERPRETATION: The long-term impacts of service change during the pandemic need full evaluation. However, widespread disruption seems to have had a profound impact on child and carer health and well-being. Service recovery needs to be specific to the individual needs of children with a disability and their families. This should be done through coproduction to ensure that service changes meet needs and are accessible and equitable.

Psychometric properties of the Viking Speech Scale-Turkish version for children with cerebral palsy aged 4-18 years based on live and video-based observation.

BACKGROUND: Speech is the most common method of communication. Video-based clinical communication evaluation is a requirement for children with speech-language impairments living in rural areas, and those who have limited mobility. AIMS: To determine the validity and reliability of the Turkish version of the Viking Speech Scale (VSS-T) via live and video-based observation for children with cerebral palsy (CP) aged 4-18 years. METHODS & PROCEDURES: A total of 142 children (mean age 8.18 ± 3.98 years; 68 female) with CP were included in this study. Their motor, communication, visual and eating-drinking function levels and comorbidities (dental, swallowing, cognitive impairments and epilepsy) were recorded. The Intelligibility in Context Scale (ICS), the Pediatric Evaluation of Disability Inventory-Social Function (PEDI-SF), and the Functional Independence Measure for Children-Communication (WeeFIM-C) were assessed to examine the concurrent validity of the VSS-T. The interrater reliability of the VSS-T was analysed between parents, physical therapists, and speech and language therapists from live and video-based observation. Intra-rater reliability was calculated from ratings made from live and video-based observations taken 3 weeks apart. OUTCOMES & RESULTS: The VSS-T was strongly related to the ICS (r = -0.830), PEDI-SF (r = -0.819), WeeFIM-C (r = -0.643), other functional classifications (r > 0.432), and the comorbidities (Cramer's V > 0.284, p < 0.001). Good to excellent interrater reliability (κw ≥ 0.838) and intra-rater reliability (intraclass correlation coefficient (ICC) = 0.848-0.995) were found between parents and therapists. CONCLUSIONS & IMPLICATIONS: Speech and language therapists, physical therapists, and parents can use the VSS-T as a valid and reliable classification system to describe speech intelligibility of 4-18-year-old children with CP. Both live and video-based observations can be used to administer the VSS-T. WHAT THIS PAPER ADDS: What is already known on the subject The English version of the VSS has been shown to be a valid and reliable tool used to classify the speech of children with CP aged 4-13 years. The scale can be administered by means of live observation of the child or based on clinicians' notes on the case by parents, SLTs, physiotherapists and paediatricians. What this paper adds to existing knowledge The VSS-T is valid and reliable for children with CP aged 4-18 years. Video-based observation is a suitable method for evaluating the VSS-T levels. The VSS-T has a moderate association with the CFCS. What are the potential or actual clinical implications of this work? The VSS-T is a valid and reliable method of categorizing the severity of motor speech impairment for Turkish children with CP in clinical research studies, registry systems or epidemiological studies. Both experienced and inexperienced therapists can use either live or video-based observation methods to administer the VSS-T. This study extended the validity and reliability of the scale in children with CP aged up to 18 years. The VSS-T is also associated with the Visual Functional Classification System (VFCS), which has been recently developed for describing the visual abilities of children with CP in daily life. In addition, the VSS-T is associated with the presence of dental, swallowing, cognitive problems and epilepsy.

Reliability and validity of the Dutch-language version of the Viking Speech Scale in children with cerebral palsy.

PURPOSE: The Viking Speech Scale is used to classify speech performance in children with cerebral palsy (CP). A Dutch-language version (VSS-NL) has recently become available. This study aimed to determine the reliability and validity of the VSS-NL and the association with motor type of CP, Gross Motor Function Classification System (GMFCS), Manual Ability Classification System (MACS) and Communication Function Classification System (CFCS). METHODS: A total of 90 children with CP, recruited throughout the Netherlands, took part in the study. VSS-NL classifications by speech and language therapists unfamiliar (SLT1) and familiar (SLT2) with the child, parents and physicians were compared. Convergent and discriminant validity were determined with the Intelligibility in Context Scale-Dutch (ICS-NL) and the Computer Based Instrument for Low Motor Language Testing (C-BiLLT). Inter- and intrarater reliability were determined by weighted Kappa (ƙw ). Validity and associations between VSS-NL and GMFCS, MACS and CFCS were determined with Spearman's coefficient. Association between VSS-NL and motor type of CP was determined with Fisher's exact test. RESULTS: Interrater reliability was excellent between SLT1-SLT2 (ƙw  = 0.93, 95% confidence interval [CI] 0.87-0.99), good between SLT1-parent (ƙw = 0.71, 95% CI 0.60-0.83), SLT1-physician (ƙw = 0.70, 95% CI 0.58-0.81), SLT2-parent (ƙw = 0.71, 95% CI 0.57-0.84), SLT2-physician (ƙw = 0.73, 95% CI 0.62-0.85) and parent-physician (ƙ = 0.72, 95% CI 0.60-0.85). Intrarater reliability was excellent for SLTs familiar and unfamiliar to the child (ƙw = 1.00, 95% CI 1.00-1.00), and very good for physicians (ƙw = 0.89, 95% CI 0.75-1.00) and parents (ƙw = 0.72, 95% CI 0.62-1.00). Convergent validity was very strong (r = -0.81, p < 0.001) and discriminant validity moderate (r = -0.56, p < 0.001). Association with motor type of CP was significant (χ2 = 27.558, p < 0.001) and strong with GMFCS (r = 0.62, p < 0.001), MACS (r = 0.63, p < 0.01) and CFCS (r = 0.69, p < 0.001). CONCLUSION: The VSS-NL is a reliable and valid system to classify speech performance in children with cerebral palsy. Classifications can be performed by SLTs, parents and physicians.

School-based allied health interventions for children and young people affected by neurodisability: a systematic evidence map.

PURPOSE: To systematically map available evidence for school-based interventions led by allied health (i.e., occupational therapy, physiotherapy, and/or speech and language therapy). MATERIALS AND METHODS: We searched for studies in pre-school, primary, secondary, or post-secondary settings, published 2004-2020. We coded study, population, and intervention characteristics. Outcomes were coded inductively, categorised, and linked to the International Classification of Functioning, Disability, and Health. RESULTS: We included 337 studies (33 countries) in an interactive evidence map. Participants were mainly pre-school and primary-aged, including individuals with neurodisability and whole-school populations. Interventions targeted wide-ranging outcomes, including educational participation (e.g., writing, reading) and characteristics of school environments (e.g., educators' knowledge and skills, peer support). Universal, targeted, and intensive interventions were reported in 21.7%, 38.9%, and 60.2% of studies, respectively. Teachers and teaching assistants delivered interventions in 45.4% and 22.6% of studies, respectively. 43.9% of studies conducted early feasibility testing/piloting and 54.9% had ≤30 participants. Sixty-two randomised controlled trials focused on intervention evaluation or implementation. CONCLUSIONS: In the United Kingdom, future research should take forward school-based allied health interventions that relate directly to agreed research priorities. Internationally, future priorities include implementation of tiered (universal, targeted, intensive) intervention models and appropriate preparation and deployment of the education workforce. IMPLICATIONS FOR REHABILITATIONAllied health professionals (occupational therapists, physiotherapists, and speech and language therapists) work in schools supporting children and young people affected by neurodisability but the content, impact, and cost-effectiveness of their interventions are not well-understood.We systematically mapped the available evidence and identified that allied health school-based interventions target highly diverse health-related outcomes and wider determinants of children and young people's health, including educational participation (e.g., literacy) and characteristics of the school environment (e.g., educators' knowledge and skills).Our interactive evidence map can be used to help stakeholders prioritise the interventions most in need of further evaluation and implementation research, including tiered models of universal, targeted, and intensive allied health support.Teachers and teaching assistants play a central role in delivering allied health interventions in schools - appropriate preparation and deployment of the education workforce should therefore be a specific priority for future international allied health research.

Advancing cluster randomised trials in children's therapy: a survey of the acceptability of trial behaviours to therapists and parents.

BACKGROUND: Randomised controlled trials of non-pharmacological interventions in children's therapy are rare. This is, in part, due to the challenges of the acceptability of common trial designs to therapists and service users. This study investigated the acceptability of participation in cluster randomised controlled trials to therapists and service users. METHODS: A national electronic survey of UK occupational therapists, physiotherapists, speech and language therapists, service managers, and parents of children who use their services. Participants were recruited by NHS Trusts sharing a link to an online questionnaire with children's therapists in their Trust and with parents via Trust social media channels. National professional and parent networks also recruited to the survey. We aimed for a sample size of 325 therapists, 30 service managers, and 60 parents. Trial participation was operationalised as three behaviours undertaken by both therapists and parents: agreeing to take part in a trial, discussing a trial, and sharing information with a research team. Acceptability of the behaviours was measured using an online questionnaire based on the Theoretical Framework of Acceptability constructs: affective attitude, self-efficacy, and burden. The general acceptability of trials was measured using the acceptability constructs of intervention coherence and perceived effectiveness. Data were collected from June to September 2020. Numerical data were analysed using descriptive statistics and textual data by descriptive summary. RESULTS: A total of 345 survey responses were recorded. Following exclusions, 249 therapists and 40 parents provided data which was 69.6% (289/415) of the target sample size. It was not possible to track the number of people invited to take the survey nor those who viewed, but did not complete, the online questionnaire for calculation of response rates. A completion rate (participants who completed the last page of the survey divided by the participants who completed the first, mandatory, page of the survey) of 42.9% was achieved. Of the three specified trial behaviours, 140/249 (56.2%) therapists were least confident about agreeing to take part in a trial. Therapists (135/249, 52.6%) reported some confidence they could discuss a trial with a parent and child at an appointment. One hundred twenty of 249 (48.2%) therapists reported confidence in sharing information with a research team through questionnaires and interviews or sharing routine health data. Therapists (140/249, 56.2%) felt that taking part in the trial would take a lot of effort and resources. Support and resources, confidence with intervention allocation, and sense of control and professional autonomy over clinical practice were factors that positively affected the acceptability of trials. Of the 40 parents, twelve provided complete data. Most parents (18/40, 45%) agreed that it was clear how trials improve children's therapies and outcomes and that a cluster randomised trial made sense to them in their therapy situation (12/29, 30%). CONCLUSIONS: Using trials to evaluate therapy interventions is, in principle, acceptable to therapists, but their willingness to participate in trials is variable. The willingness to participate may be particularly influenced by their views related to the burden associated with trials, intervention allocation, and professional autonomy.

Prevalence of congenital heart defects in Europe, 2008-2015: A registry-based study.

BACKGROUND: The total prevalence of congenital heart defects (CHDs) varies by populations and over time. Studies that examine trends in the prevalence of CHD in different regions may shed light on our understanding of the occurrence of CHD and the impact of different risk factors. OBJECTIVES: To examine trends in total and live birth prevalence of nonsyndromic CHD in Europe between the years 2008 and 2015 and to investigate if the decreasing trend reported by previous studies is continuing. METHODS: Cases of CHD delivered between January 1, 2008 and December 31, 2015 notified to 25 population-based EUROCAT (European Surveillance of Congenital Anomalies) registries in 14 countries, formed the population-based case-series. Prevalence (total/live) rates and 95% confidence intervals were calculated as the number of cases per 10,000 births (live and stillbirths). Time trends in prevalence of all nonsyndromic CHDs and for three CHD severity groups (very severe, severe, and less severe) were plotted using a Poisson regression multilevel approach. RESULTS: The total prevalence of nonsyndromic CHD was 57.1 per 10,000 births (live births and stillbirths) for the 8-year period and remained stable across the three CHD severity groups while the live birth prevalence was 60.2 per 10,000 births. There was considerable variation in the reported total CHD prevalence and the direction of trends by registry. A decreasing prevalence of CHD was observed for the Norway and England/Wales registries, whereas the CHD prevalence increased for registries in Italy and Croatia. CONCLUSIONS: The total prevalence of CHD in Europe between the years 2008 and 2015 remained stable for all CHD and across the three CHD severity groups. The decreasing trend reported by previous studies has not continued. However, we found significant differences in the total and live birth prevalence by registry.

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.

Mini-EDACS: Development of the Eating and Drinking Ability Classification System for young children with cerebral palsy.

AIM: To develop and test Mini-EDACS to describe developing eating and drinking abilities of children with cerebral palsy (CP) aged between 18 and 36 months. METHOD: The existing Eating and Drinking Ability Classification System (EDACS) was modified to define Mini-EDACS content. Mini-EDACS was developed in three stages: (1) EDACS was modified after application to videos of standardized feeding evaluations of children with CP aged 18 to 36 months (n = 130); (2) refined content and validity of Mini-EDACS was established through an international Delphi survey; (3) interobserver reliability was assessed by comparing Mini-EDACS levels assigned by speech and language therapists (SaLTs) from video data and parent report. RESULTS: Mini-EDACS provides age-appropriate descriptions for children aged 18 to 36 months with CP. Eighty-nine stakeholders participated in the Delphi survey; required levels of agreement were met after one round (i.e. >80% agreement). Thirteen SaLTs completed paired ratings from 43 video recordings: absolute agreement was 58% (kappa 0.43; intraclass correlation coefficient 0.78; 95% confidence interval 0.63-0.87). INTERPRETATION: Mini-EDACS provides a valid system for classifying eating and drinking performance of children with CP under 3 years old. Results suggest moderate agreement and good reliability when rating Mini-EDACS levels from video recordings of young children with CP.

Oromotor dysfunction in minimally verbal children with cerebral palsy: characteristics and associated factors.

AIM: To explore the characteristics and associated factors of oromotor dysfunction in minimally verbal children with cerebral palsy (CP) aged five to six years, recruited from a population-based registry. METHODS: Twenty children with CP who were minimally verbal completed a standardised, observational oromotor assessment. Linear regression analyses examined the relationship between oromotor dysfunction and potential associated factors (e.g., fine and gross motor function, communication, and feeding). RESULTS: Oromotor dysfunction affected every participant and was identified in all structures examined (i.e., face, jaw, lips, and tongue). Oromotor movements showed little dissociation among jaw, lip, and tongue movements. Oromotor dysfunction was univariately associated with the Manual Ability Classification System levels IV-V (p = 0.001), reduced communication skills (p = 0.002), and a prolonged eating duration (>45 min) (p = 0.006), even when non-verbal cognition served as a covariate. INTERPRETATION: Oromotor dysfunction was highly prevalent in our sample of minimally verbal children with CP, having significant functional impacts on feeding and communication. Findings suggest that fine motor function (i.e., Manual Ability Classification System levels IV-V) is a stronger predictor than gross motor function for identifying children with CP who are minimally verbal and at risk of oromotor dysfunction. IMPLICATIONS FOR REHABILITATIONOromotor dysfunction was highly prevalent in our sample of minimally verbal children with cerebral palsy.Severe fine motor impairment strongly predicted oromotor dysfunction, indicating that fine motor function may provide an early indicator of impaired oromotor function for this clinical population.Robust, standardised measures of motor speech-related oromotor development suitable for children with cerebral palsy who are minimally verbal are lacking.Until such a measure is developed, formal evaluation may be achieved via oral motor assessments standardised for typically developing children, with the caveat one must interpret the results with caution.

Determinants of participation and quality of life of young adults with cerebral palsy: longitudinal approach and comparison with the general population - SPARCLE 3 study protocol.

BACKGROUND: Effective inclusion in society for young people with disabilities is increasingly seen as generating opportunities for self-development, and improving well-being. However, significant barriers remain in the vast majority of activities meaningful for young adults. Research argues that various personal (disabilities, health) and environmental (access to the resources needed, accessible environment, discrimination, lack of personal economic independence) factors contribute to limited participation. However, previous studies conducted in young people with cerebral palsy (CP) mainly investigated the transition period to adulthood, and did not fully consider the whole range of impairment severity profiles or environmental barriers. In this study, we will use the follow-up of the SPARCLE cohort and a comparison group from the general population (1) to investigate the impact of the environment on participation and quality of life of young adults with CP, (2) to determine predictors of a successful young adulthood in educational, professional, health and social fields, (3) to compare quality of life and frequency of participation in social, work and recreational activities with the general population, (4) to document on participation and quality of life in those with severe disabilities. METHODS: The SPARCLE3 study has a combined longitudinal and cross-sectional design. Young adults with CP aged 22 to 27 years in 6 European regions previously enrolled in the SPARCLE cohort or newly recruited will be invited to self-complete a comprehensive set of questionnaires exploring participation (daily life and discretionary activities), health-related quality of life, body function, personal factors (health, personal resources), and contextual factors (availability of needed environmental items, family environment, services provision) during home visits supervised by trained researchers. Proxy-reports or adapted questionnaires will be used for those with the most severe impairments. The recruitment of a large group from the general population (online survey) will enable to identify life areas where the discrepancies between young people with CP and their able-bodied peers are the most significant. DISCUSSION: This study will help identify to what extent disabilities and barriers in environment negatively affect participation and quality of life, and how previous valued experiences during childhood or adolescence might modulate these effects.

Social participation to support good mental health in neurodisability.

BACKGROUND: Young people with neurodisability experience lower levels of mental wellbeing and are at increased risk of mental illness compared with their non-disabled peers. Social participation is recognized as a protective factor against mental illness and a potential pathway to support better mental wellbeing in neurodisability. METHOD: This co-design study involved young people, parents and clinicians. First, possible interventions were identified through a rapid systematic evidence review. Any study designs were considered, which included people with a neurodisability aged 0-18 years, which evaluated a therapy intervention with social participation and mental health outcomes. Titles and abstracts were screened by two reviewers, from the included studies data were extracted and then presented using written summaries. Second, the summaries were discussed and prioritized in stakeholder groups with young people, parents and clinicians. Groups were audio recorded and framework analysis was used to identify and specify intervention elements and their delivery. RESULTS: The evidence review identified 13,870 records, from which 43 were included. These records were published 1994-2017 and reported studies with 4-249 participants aged 16 months-18 years with a range of neurodisabilities. Five intervention approaches (social skills training, arts, sports, technology and play) were identified from the review. Two themes emerged from the stakeholder groups: intervention in the real world, feeling judged and feeling safe. The groups prioritized an intervention in real-world social leisure contexts (i.e. existing clubs and groups) using nine key intervention elements (e.g. feedback and positive verbal reinforcement) delivered by club leaders trained by healthcare professionals using five intervention procedures (e.g. a manual and video training). CONCLUSION: This study has identified core elements of social participation interventions that may improve mental health outcomes in young people with neurodisability, which should now be tested.

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.

A randomised controlled feasibility study of interpersonal art psychotherapy for the treatment of aggression in people with intellectual disabilities in secure care.

BACKGROUND: Rates of aggression in inpatient secure care are higher than in other psychiatric inpatient settings. People with intellectual disabilities in secure care require adapted psychological treatments. Interpersonal art psychotherapy incorporates the use of creative art making approaches by participants, thus reducing sole reliance upon verbal interactions during psychotherapy for people who may have communication difficulties. During interpersonal art psychotherapy, participants are individually supported by their therapist to consider how they conduct relationships. This includes the influence and impact of interpersonal issues resulting in repeated patterns of conflict. The key feasibility objectives were to assess recruitment and retention rates, follow-up rates and trial procedures such as randomisation, allocation and identifying any practical or ethical problems. In addition, a preliminary 'signal' for the intervention was considered and an indicative sample size calculation completed. The acceptability of a potential third trial arm attentional control condition, mindful colouring-in, was assessed using four single-case design studies and a UK trial capacity survey was conducted. METHODS: Adult patients with intellectual disabilities in secure care were recruited and randomised to either interpersonal art psychotherapy or delayed treatment in this multi-site study. Outcomes were assessed using weekly observations via the Modified Overt Aggression Scale and a range of self-report measures. Within study reporting processes, qualitative interviews and a survey were completed to inform trial feasibility. RESULTS: Recruitment procedures were successful. The target of recruiting 20 participants to the trial from multiple sites was achieved within 8 months of the study opening. All participants recruited to the treatment arm completed interpersonal art psychotherapy. Between-group differences of interpersonal art psychotherapy versus the delayed treatment control showed a 'signal' effect-size of .65 for total scores and .93 in the verbal aggression sub-scale. There were no amendments to the published protocol. The assessment of key feasibility objectives were met and the trial procedures were acceptable to all involved in the research. CONCLUSION: This study suggested that a randomised controlled trial of interpersonal art psychotherapy is acceptable and feasible. TRIAL REGISTRATION: ISRCTN14326119 (Retrospectively Registered).

Communication disorders in young children with cerebral palsy.

AIM: To test the prediction of communication disorder severity at 5 years of age from characteristics at 2 years for children with cerebral palsy (CP) whose communication is giving cause for concern. METHOD: In this cohort study, 77 children (52 males; 25 females) with communication difficulties and CP were visited at home at 2 (mean 2y 4mo; SD 3mo) and 5 (mean 5y 5mo; SD 4mo) years of age. Information on the type and distribution of motor disorder, seizures, gross and fine motor function, hearing, and vision were collected from medical notes. Non-verbal cognition, language comprehension, language expression, spoken vocabulary, and methods of communication were assessed directly at age 2 years. At 5 years, communication and speech function were rated using the Communication Function Classification System (CFCS), Functional Communication Classification System (FCCS), and Viking Speech Scale (VSS). RESULTS: In multivariable regression models, CP type, Gross Motor Function Classification System level, vision, the amount of speech understood by strangers, non-verbal cognition, and number of consonants produced at age 2 years predicted the CFCS level at age 5 years (R2 =0.54). CP type, Manual Ability Classification System level, amount of speech understood, vision, and number of consonants predicted the FCCS level (R2 =0.49). CP type, amount of speech understood by strangers, and number of consonants predicted the VSS level (R2 =0.50). INTERPRETATION: Characteristics at 2 years of age predict communication and speech performance at 5 years, and should inform referral to speech and language therapy.

Speech in children with cerebral palsy.

AIM: To examine the frequency, characteristics, and factors associated with speech delay and disorder in a community sample of children with cerebral palsy (CP). METHOD: Participants were 84 children (37 females, 47 males; aged between 4y 11mo-6y 6mo) with CP identified through a population-based registry. Speech and oromotor function were systematically evaluated to provide a differential diagnosis of articulation, phonological, and motor speech disorders. RESULTS: In total, 82% (69/84) of participants had delayed or disordered speech production, including minimally verbal presentations (n=20). Verbal participants (n=64) presented with dysarthria (78%), articulation delay or disorder (54%), phonological delay or disorder (43%), features of childhood apraxia of speech (CAS) (17%), or mixed presentations across these conditions. Speech intelligibility was poorest in those with dysarthria and features of CAS. Speech delay or disorder in verbal participants was associated with language impairment (p=0.002) and reduced health-related quality of life (p=0.04) (Fisher's exact test). Poorer speech accuracy (i.e. lower percentage consonants correct) correlated with greater impairments in both language (p<0.001) and oromotor function (p<0.001) (Spearman's test). INTERPRETATION: The speech profile of children with CP is characterized by impairment at multiple levels of speech production (phonetic, cognitive-linguistic, neuromuscular execution, and high-level planning/programming), highlighting the importance of a personalized differential diagnosis informing targeted treatment.

Views of children with cerebral palsy and their parents on the effectiveness and acceptability of intensive speech therapy.

Purpose: To understand children and parents' views of the effectiveness and acceptability of intensive dysarthria therapy.Materials and Methods: Twenty-two children with cerebral palsy and dysarthria joined a pilot RCT comparing intensive therapy and usual care. Children (n = 11) allocated to dysarthria therapy comprising three 40-minute sessions per week for six weeks and their parents (n = 11) were interviewed two weeks before and six weeks after therapy. Interviews were transcribed verbatim and analysed thematically.Results: Analysis revealed five themes: Motivations, My new voice; The new me; I can do more; Success rooted in therapy design. Children had received little therapy for speech and were keen to improve intelligibility. Overall, therapy was viewed as effective. Participants described changes in children's speech production, which they associated with increased speech intelligibility. Children were described as more confident following the therapy, to have more successful conversations, with a wider range of partners in more environments, thereby increasing their social participation. The programme was viewed as acceptable, despite its intensity, due to the short term commitment and wider benefits for the child. Parents valued the organised structure and individualisation of the programme and inclusion in the therapy process.Conclusion: Families found the intervention acceptable and effective. A definitive trial of its clinical effectiveness is warranted.Implications for rehabilitationChildren with cerebral palsy who have dysarthria and their parents reported that intensive speech therapy focussing on creating a stronger voice and a steady speech rate increased the clarity of children's voice and the intelligibility of their speech.Therapy may have additional benefits for children's self-confidence and social participation.The programme of therapy comprising three sessions per week for six weeks was seen as manageable by families in view of the results achieved.