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Background The FEEDS (Focus on Early Eating, Drinking and Swallowing) study focused on interventions used to improve feeding for children with neurodisability and eating, drinking and swallowing difficulties (EDSD), and the outcomes viewed as important by healthcare professionals (HPs) and parent carers. The FEEDS Toolkit was created subsequently as an intervention decision aid to be used collaboratively by parent carers and HPs. This study aimed to inform on current intervention practices and influence toolkit design by ascertaining whether specific intervention use varied by a child’s main diagnosis and by specific HP role.Methods FEEDS survey data were grouped by child’s main diagnosis and HP role. Main diagnoses included autism spectrum disorder (ASD) n=183; Down syndrome (DS) n=69; cerebral palsy (CP) n=30). HPs included were speech and language therapists (SLT) n=131; occupational therapists (OT) n=63; physiotherapists (PT) n=57; paediatricians n=50; dieticians n=40; nurses n=32 and health visitors n=14.Results Most interventions were used commonly across diagnoses. However, some interventions were used more commonly with specific conditions, for example, positioning (CP 85%, DS 70%, ASD 23%, strategies/programmes aimed at changing behaviour at mealtimes (ASD 52%, CP 8%, DS 11%); visual supports (ASD 58%, CP 0%, DS 21%). HPs reported using a broad range of interventions, SLTs (mean=13.9), dieticians (12.3), OTs (12.7) and paediatricians (11.1). There was overlap between intervention use and HP role, for example, positioning (100% PT, 97% SLT, 94% OT, 73% paediatricians and 69% nurses).Conclusions Interdisciplinary working is common when managing EDSD, with all HP types using multiple interventions. A child’s main diagnosis does not substantially influence intervention use, and the individual context of each child requires consideration in intervention selection. Study findings have supported development of the FEEDS Toolkit for use in feeding services.

Objectives To understand how health, education and social care services for disabled children changed during the COVID-19 pandemic, what did or did not work well and what the impacts of service changes were on both professionals and families.Design Qualitative study using semistructured interviews.Setting Telephone and video call interviews and focus groups with professionals working in one of five local authority areas in England.Participants 78 health, education and social care professionals working with children in one of five local authority areas in England.Results There was a significant disruption to services and reduced contact with families during the early stages of the pandemic; nevertheless, professionals were able to reflect on innovative ways they interacted with and sought to support and maintain health, education and social care provision to disabled children and their families. As waitlists have substantially increased, this and the longevity of the pandemic were perceived to have had negative consequences for staff health and well-being, the health and psychosocial outcomes of children and young people, and their parent carers.Conclusions Key learning from this study for service recovery and planning for future emergencies is the need to be able to identify disabled children, classify their level of need and risk, assess the impact of loss of services and maintain clear communication across services to meet the needs of disabled children. Finally, services need to work collaboratively with families to develop child-centred care to strengthen resilience during service disruption.

AbstractScientific literacy requires students to generalise their scientific understanding to contexts beyond the classroom and engage in effective communication. An interdisciplinary approach with the curriculum areas of science and English could address the increasingly complex and multidisciplinary needs of future citizens. This scoping review targets research concerning written argumentation, which is typically positioned as an educational goal or demonstration of learning in English and science education. The systematic literature search process yielded a total of 260 research outputs, with a total of approximately 54,233 participants included across all outputs. Research themes in science written argumentation research were frequently related to content, scaffolding, and instructional style, while English written argumentation research had a more even distribution of research interests, showing a slight inclination for student characteristics and genre. It is clear that argumentation in science education should move beyond a purely objectivist emphasis on facts to a fuller consideration of perspectives, norms, and rhetorical features in English argumentation to further improve the collective scientific literacy of our learners.

Abstract Background Parent carers of disabled children are at increased risk of physical and mental health problems. The Healthy Parent Carers (HPC) programme is a manualised peer‐led group‐based programme that aims to promote parent carer health and wellbeing. Previously, the programme had been delivered in person, with recruitment and delivery managed in a research context. This study explored implementation by two delivery partner organisations in the United Kingdom. Facilitator Training and Delivery Manuals were modified for online delivery using Zoom due to COVID‐19. Methods The study methodology utilised the Replicating Effective Programs framework. A series of stakeholder workshops informed the development of the Implementation Logic Model and an Implementation Package. After delivering the programme, delivery partner organisations and facilitators participated in a workshop to discuss experiences of implementing the programme. A wider group of stakeholders, including commissioners, Parent Carer Forums and charity organisations representatives and researchers subsequently met to consider the sustainability and potential barriers to delivering the programme outside the research context. Results This study explored implementation by two delivery partner organisations in the United Kingdom that were able to recruit facilitators, who we trained, and they recruited participants and delivered the programme to parent carers in different localities using Zoom. The co‐created Implementation Logic Model and Implementation Package were subsequently refined to enable the further roll‐out of the programme with other delivery partner organisations. Conclusions This study provides insight and understanding of how the HPC programme can be implemented sustainably outside of the research context. Further research will evaluate the effectiveness of the programme and refine the implementation processes. Patient and Public Contribution Parent carers, delivery partner organisation staff and service commissioners were consulted on the design, delivery and reporting of the research.

Thermoplastic composite organosheets (OSs) are increasingly recognized as a viable solution for automotive and aerospace structures, offering a range of benefits including cost-effectiveness through high-rate production, lightweight design, impact resistance, formability, and recyclability. This study examines the impact response, post-impact strength evaluation, and hot-pressing repair effectiveness of woven glass fiber nylon composite OSs across varying impact energy levels. Experimental investigations involved subjecting composite specimens to impact at varying energy levels using a drop-tower test rig, followed by compression-after-impact (CAI) tests. The results underscore the exceptional damage tolerance and improved residual compressive strength of the OSs compared to traditional thermoset composites. This enhancement was primarily attributed to the matrix’s ductility, which mitigated transverse crack propagation and significantly increased the amount of absorbed energy. To mitigate impact-induced damage, a localized hot-pressing repair approach was developed. This allowed to restore the post-impact strength of the OSs to pristine levels for impact energies below 40 J and by 83.6% for higher impact energies, when OS perforation was observed. The measured levels of post-repair strength demonstrate a successful restoration of OS strength over a wide range of impact energies, and despite limitations in achieving complete strength recovery above 40 J, hot-pressing repair emerges as a promising strategy for ensuring the longevity of thermoplastic composites through repairability.

An experimental investigation of interlaminar toughness for post-cured through-thickness reinforcement (PTTR) skin–stringer sub-element is presented. The improvement in the crack resistance capability of skin–stringer samples was shown through experimental testing and finite element analysis (FEA) modeling. The performance of PTTR was evaluated on a pristine and initial-disbond of the skin–stringer specimen. A macro-scale pin–spring modeling approach was employed in FEA using a non-linear spring to capture the pin failure under the mixed-mode load. The experimental results showed a 15.5% and 20.9% increase in strength for the pristine-PTTR and initial-disbond PTTR specimens, respectively. The modeling approach accurately represents the overall structural response of PTTR laminate, including stiffness, adhesive strength, crack extension scenarios and progressive pin failure modes. This modeling approach can be beneficial for designing damage-tolerant structures by exploring various PTTR arrangements for achieving improved structural responses.

Objective Describe families’ experiences of interventions to improve continence in children and young people with neurodisability, and health professionals’ and school and social care staff’s perspectives regarding factors affecting intervention use.Design Four online surveys were developed and advertised to parent carers, young people with neurodisability, health professionals and school and social care staff, via societies, charities, professional contacts, schools, local authorities, and national parent carer and family forums, who shared invitations with their networks. Survey questions explored: difficulties helping children and young people use interventions; acceptability of interventions and waiting times; ease of use and availability of interventions, and facilitators and barriers to improving continence.Results 1028 parent carers, 26 young people, 352 health professionals and 202 school and social care staff registered to participate. Completed surveys were received from 579 (56.3%) parent carers, 20 (77%) young people, 193 (54.8%) health professionals, and 119 (58.9%) school and social care staff. Common parent carer-reported difficulties in using interventions to help their children and young people to learn to use the toilet included their child’s lack of understanding about what was required (reported by 337 of 556 (60.6%) parent carers who completed question) and their child’s lack of willingness (343 of 556, 61.7%). Almost all (142 of 156, 91%) health professionals reported lack of funding and resources as barriers to provision of continence services. Many young people (14 of 19, 74%) were unhappy using toilet facilities while out and about.Conclusions Perceptions that children lack understanding and willingness, and inadequate facilities impact the implementation of toileting interventions for children and young people with neurodisability. Greater understanding is needed for children to learn developmentally appropriate toileting skills. Further research is recommended around availability and acceptability of interventions to ensure quality of life is unaffected.

The etiology of schizophrenia (SCZ) is multifactorial, and depending on a host of genetic and environmental factors. Two putative SCZ susceptibility genes, Disrupted-in-Schizophrenia-1 (DISC1) and reelin (RELN), interact at a molecular level, suggesting that combined disruption of both may lead to an intensified SCZ phenotype. To examine this gene–gene interaction, we produced a double mutant mouse line. Mice with heterozygous RELN haploinsufficiency were crossed with mice expressing dominant-negative c-terminal truncated human DISC1 to produce offspring with both mutations (HRM/DISC1 mice). We used an array of behavioral tests to generate a behavioral phenotype for these mice, then examined the prefrontal cortex and hippocampus using western blotting and immunohistochemistry to probe for SCZ-relevant molecular and cellular alterations. Compared to wild-type controls, HRM/DISC1 mice demonstrated impaired pre-pulse inhibition, altered cognition, and decreased activity. Diazepam failed to rescue anxiety-like behaviors, paradoxically increasing activity in HRM/DISC1 mice. At a cellular level, we found increased α1-subunit containing GABA receptors in the prefrontal cortex, and a reduction in fast-spiking parvalbumin positive neurons. Maturation of adult-born neurons in the hippocampus was also altered in HRM/DISC1 mice. While there was no difference in the total number proliferating cells, more of these cells were in immature stages of development. Homozygous DISC1 mutation combined with RELN haploinsufficiency produces a complex phenotype with neuropsychiatric characteristics relevant to SCZ and related disorders, expanding our understanding of how multiple genetic susceptibility factors might interact to influence the variable presentation of these disorders.

Abstract Background In paediatric epilepsy, the evidence of effectiveness of antiseizure treatment is inconclusive for some types of epilepsy. As with other paediatric clinical trials, researchers undertaking paediatric epilepsy clinical trials face a range of challenges that may compromise external validity Main body In this paper, we critically reflect upon the factors which impacted recruitment to the pilot phase of a phase IV unblinded, randomised controlled 3×2 factorial trial examining the effectiveness of two antiseizure medications (ASMs) and a sleep behaviour intervention in children with Rolandic epilepsy. We consider the processes established to support recruitment, public and patient involvement and engagement (PPIE), site induction, our oversight of recruitment targets and figures, and the actions we took to help us understand why we failed to recruit sufficient children to continue to the substantive trial phase. The key lessons learned were about parent preference, children’s involvement and collaboration in decision-making, potential and alternative trial designs, and elicitation of stated preferences pre-trial design. Despite pre-funding PPIE during the trial design phase, we failed to anticipate the scale of parental treatment preference for or against antiseizure medication (ASMs) and consequent unwillingness to be randomised. Future studies should ensure more detailed and in-depth consultation to ascertain parent and/or patient preferences. More intense engagement with parents and children exploring their ideas about treatment preferences could, perhaps, have helped predict some recruitment issues. Infrequent seizures or screening children close to natural remission were possible explanations for non-consent. It is possible some clinicians were unintentionally unable to convey clinical equipoise influencing parental decision against participation. We wanted children to be involved in decisions about trial participation. However, despite having tailored written and video information to explain the trial to children we do not know whether these materials were viewed in each consent conversation or how much input children had towards parents’ decisions to participate. Novel methods such as parent/patient preference trials and/or discrete choice experiments may be the way forward. Conclusion The importance of diligent consultation, the consideration of novel methods such as parent/patient preference trials and/or discrete choice experiments in studies examining the effectiveness of ASMs versus no-ASMs cannot be overemphasised even in the presence of widespread clinician equipoise.

Targeted therapies such as venetoclax (VEN) (Bcl-2 inhibitor) have revolutionized the treatment of chronic lymphocytic leukemia (CLL). We previously reported that persister CLL cells in treated patients overexpress multiple antiapoptotic proteins and display resistance to proapoptotic agents. Here, we demonstrated that multidrug-resistant CLL cells in vivo exhibited apoptosis restriction at a pre-mitochondrial level due to insufficient activation of the Bax and Bak (Bax/Bak) proteins. Co-immunoprecipitation analyses with selective BH domain antagonists revealed that the pleiotropic proapoptotic protein (Bim) was prevented from activating Bax/Bak by “switching” interactions to other upregulated antiapoptotic proteins (Mcl-1, Bcl-xL, Bcl-2). Hence, treatments that bypass Bax/Bak restriction are required to deplete these resistant cells in patients. Protein phosphatase 2A (PP2A) contributes to oncogenesis and treatment resistance. We observed that small-molecule activator of PP2A (SMAP) induced cytotoxicity in multiple cancer cell lines and CLL samples, including multidrug-resistant leukemia and lymphoma cells. The SMAP (DT-061) activated apoptosis in multidrug-resistant CLL cells through induction of mitochondrial permeability transition pores, independent of Bax/Bak. DT-061 inhibited the growth of wild-type and Bax/Bak double-knockout, multidrug-resistant CLL cells in a xenograft mouse model. Collectively, we discovered multidrug-resistant CLL cells in patients and validated a pharmacologically tractable pathway to deplete this reservoir.

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

Introduction Sleep and epilepsy have an established bidirectional relationship yet only one randomised controlled clinical trial has assessed the effectiveness of behavioural sleep interventions for children with epilepsy. The intervention was successful, but was delivered via face-to-face educational sessions with parents, which are costly and non-scalable to population level. The Changing Agendas on Sleep, Treatment and Learning in Epilepsy (CASTLE) Sleep-E trial addresses this problem by comparing clinical and cost-effectiveness in children with Rolandic epilepsy between standard care (SC) and SC augmented with a novel, tailored parent-led CASTLE Online Sleep Intervention (COSI) that incorporates evidence-based behavioural components.Methods and analyses CASTLE Sleep-E is a UK-based, multicentre, open-label, active concurrent control, randomised, parallel-group, pragmatic superiority trial. A total of 110 children with Rolandic epilepsy will be recruited in outpatient clinics and allocated 1:1 to SC or SC augmented with COSI (SC+COSI). Primary clinical outcome is parent-reported sleep problem score (Children’s Sleep Habits Questionnaire). Primary health economic outcome is the incremental cost-effectiveness ratio (National Health Service and Personal Social Services perspective, Child Health Utility 9D Instrument). Parents and children (≥7 years) can opt into qualitative interviews and activities to share their experiences and perceptions of trial participation and managing sleep with Rolandic epilepsy.Ethics and dissemination The CASTLE Sleep-E protocol was approved by the Health Research Authority East Midlands (HRA)–Nottingham 1 Research Ethics Committee (reference: 21/EM/0205). Trial results will be disseminated to scientific audiences, families, professional groups, managers, commissioners and policymakers. Pseudo-anonymised individual patient data will be made available after dissemination on reasonable request.Trial registration number ISRCTN13202325.

Abstract Background Parent carers of children with special educational needs or disability are at higher risk of poor mental and physical health. The need for a tailored, peer-led group programme was raised by parent carers, who co-developed the Healthy Parent Carers programme with researchers. This study aimed to test the feasibility of programme delivery in community settings, and the feasibility and acceptability of a randomised controlled trial design. Methods Participants were individually randomised with concealed allocation to a structured group programme and access to online resources (intervention), or access to the online resources only (control). Measures of wellbeing and secondary and economic outcomes were collected before randomisation, immediately post-intervention, and 6 months post-intervention. Descriptive statistics on recruitment and attrition, demographics, attendance, and fidelity of intervention delivery were analysed with feedback on the acceptability of the trial design. Results One hundred and ninety-three parent carers expressed an interest in taking part. Ninety-two participants recruited from across six sites were randomised (47 intervention, 45 control). Lead and assistant facilitators were trained and delivered the group sessions. Sixteen (34%) participants in the intervention arm did not attend any sessions, and attendance varied across sites and sessions. One participant withdrew post-randomisation, and 83 (90%) participants completed outcome measures at the six-month follow-up. Conclusions The study demonstrated that it was feasible to deliver the programme in community settings. The number of parent carers who expressed interest signifies the need for such a programme and the feasibility of recruiting to a definitive trial. Loss to follow-up was low. Further research is needed to explore ways to reduce barriers to participation in person and assess the feasibility and acceptability of programme content and delivery for more ethnically diverse groups, and potentially using interpreters. Given the Covid-19 pandemic and delivery format feedback, there is also a need to investigate remote or blended delivery strategies. Although the results indicate that a definitive trial is feasible, programme impact would be strengthened through exploration of these uncertainties. Trial registration ISRCTN, ISRCTN15144652 , registered on 25 October 2018, ClinicalTrials.gov , NCT03705221 , registered on 15 October 2018.

Plain English summary Background Patient and public involvement means researchers working with members of the public, patients or carers to jointly plan and carry out research. Aim This article is written by members of three involvement groups, and the university employees that they work with. We wanted to jointly reflect on what enables our collaborative work, and what the challenges are for everyone involved. What we did and how we did it We wanted to establish what the literature defines as ‘good’ public involvement and compare this with processes and practices in our involvement groups. We therefore carried out a literature review and each group met separately to discuss what characterises good involvement, and what the challenges are. From these discussions we developed a set of descriptions about each group. We compared the literature review findings with what came out of the discussions within the involvement groups. Findings Some of the involvement principles from the literature were similar to the priorities of the involvement groups. In addition, the groups identified characteristics of ‘good’ involvement practice that were not reported in the literature: passion and enthusiasm, informal and welcoming meeting spaces, and opportunities to share lived experiences. In this article we present examples of how principles for good involvement are practiced in these groups, and difficulties we have experienced. Abstract Background Patient and public involvement is important for producing relevant and accessible health research. Evidence of impact from involvement is growing, but there is also a need for research on how to create conditions for meaningful collaborations between researchers and public advisers. Objective We report on a co-produced self-reflective evaluation of involvement practices in three UK research programmes. Methods A structured review identified research-based principles for ‘good’ public involvement in research. In parallel, members of three involvement groups co-developed statements on how the groups work, and enablers and challenges to collaborative research. The author team analysed these statements using the findings from the review. Results We identified 11 international articles reporting research-based principles for involvement published between 2013 and 2017. We identified five ‘values’ and seven ‘practice principles’ for ‘good’ involvement. There was convergence between these principles and the priorities of the involvement groups. But the groups also identified additional good involvement practice that were not reported by the literature: passion, enthusiasm, informal and welcoming meeting spaces, and opportunities to share lived experiences. We present examples of how principles for good involvement are practiced in these groups, and highlight principles that have been challenging to implement. Conclusions Ongoing appraisal of public involvement is crucial. We present a process for self-evaluation, illuminate what ‘good’ means to researchers and public advisers involved in research, and identify areas for improvement. We conclude that provision of resources that enable support to public advisers in turn enable universities and research teams to implement other principles of good involvement.

Abstract Graph kernels have become an established and widely-used technique for solving classification tasks on graphs. This survey gives a comprehensive overview of techniques for kernel-based graph classification developed in the past 15 years. We describe and categorize graph kernels based on properties inherent to their design, such as the nature of their extracted graph features, their method of computation and their applicability to problems in practice. In an extensive experimental evaluation, we study the classification accuracy of a large suite of graph kernels on established benchmarks as well as new datasets. We compare the performance of popular kernels with several baseline methods and study the effect of applying a Gaussian RBF kernel to the metric induced by a graph kernel. In doing so, we find that simple baselines become competitive after this transformation on some datasets. Moreover, we study the extent to which existing graph kernels agree in their predictions (and prediction errors) and obtain a data-driven categorization of kernels as result. Finally, based on our experimental results, we derive a practitioner’s guide to kernel-based graph classification.

Abstract Background Parent carers of disabled children are at increased risk of mental and physical health problems. They often experience challenges to maintaining good health which have implications for their well-being and their ability to care for their children. In response to these needs, researchers and parent carers developed the Healthy Parent Carers (HPC) programme. It is a peer-led, group-based intervention that promotes behaviours associated with health and well-being. The aims of this trial are to assess the acceptability of the HPC programme and the feasibility of its delivery in the community and to assess the feasibility and acceptability of the design of the definitive trial to evaluate the programme’s effectiveness and cost-effectiveness. Methods We will establish six research sites and train facilitators to deliver the manualised intervention. Parent carers of children with special educational needs and disabilities will be individually randomised, stratified by group delivery site, to either take part in a group programme and online resources (intervention) or to receive access to the online resources only (control). Measures of mental health; well-being; health-related quality of life; health behaviours; patient activation; protective factors such as resilience, social connections, and practical support; and use of health care, social care, and wider societal resources will be collected before randomisation (baseline), immediately post-intervention, and 6 months later. Recruitment of participants, adherence to the programme, and the dose received will be assessed. Group sessions will be audio-recorded to evaluate the fidelity of delivery and participant engagement. Participants’ and facilitators’ feedback on the programme content and delivery, their experience, and the acceptability of the outcome measures and trial design will be collected through feedback forms, interviews, and focus groups. Discussion This trial will assess whether the programme delivery and evaluative trial design are feasible, to inform whether to progress to a definitive randomised controlled trial to test the effectiveness and cost-effectiveness of the Healthy Parent Carers programme. Trial registration ISRCTN, ISRCTN151144652, registered on 25 October 2018; ClinicalTrials.gov, NCT03705221, registered on 15 October 2018.

Background: Children and young people with neurodisability often need help to achieve socially acceptable bladder and bowel control. Approaches vary depending on whether or not the impairment results from spinal cord pathology that impairs motor control and sensation of the bladder and bowel. Currently, there is uncertainty about which interventions are effective. Objective: The objective was to summarise the available evidence on and current practice for improving continence in children and young people with neurodisability. Design: A systematic review of the effectiveness, cost-effectiveness and factors that modify intervention implementation, alongside a cross-sectional, online survey of current practice with health professionals, parent carers, school and care staff and young people with neurodisability. Results: Twelve databases were searched in the review, resulting in 5756 references; 71 studies (72 papers) were included in the analyses. Most of the evidence was for children with spinal cord pathology, which involved evaluations of pharmacological approaches and surgical techniques, whereas the evidence pertaining to those with non-spinal-cord-related pathology tended to be for behavioural interventions. The methodological quality of studies was rated as being moderate to poor. There were three robust qualitative studies about the experience of continence among children with spinal cord pathology. We found substantial heterogeneity across the interventions that we evaluated in terms of quality, study design and outcomes measured. No economic studies were found. The results were synthesised narratively and reported in text and tables. We did not find any eligible studies evaluating interventions using toilet and clothing adaptations in the review, although the survey highlighted that these types of interventions are frequently used and considered. In total, 949 people responded to the survey: 202 health professionals, 605 parent carers, 122 school and social care staff, and 20 young people. The survey results illustrated the different roles that professionals have in improving continence, highlighting the importance of a multidisciplinary approach to supporting children and young people and their families. Clinicians employ a range of assessments and interventions to improve continence or independent toileting, depending on the needs of the child. Limitations: Quantitative studies in the review were not methodologically robust. The survey had a risk of response bias. Conclusions: Our research found a dearth of good-quality evidence for many of the interventions currently in use, and no evidence of experiences of implementing interventions for children with non-spinal-cord-related pathology. There was also no evidence of cost-effectiveness of any of the interventions. Future work: There is a need to involve young people and families in the design of high-quality evaluative research for interventions that aim to improve continence. This is especially the case for children with autism and learning disability, who have been neglected in previous evaluative and qualitative research. We recommend better training for health, education and care professionals about toileting, informed by evidence and the lived experiences of children and their families. We recommend a joined-up multidisciplinary and holistic approach to improving continence to maximise independence, dignity and comfort. It is vital that children and young people with neurodisability have early access to regular, integrated assessment of their bladder and bowel health, and are fully supported with appropriate personalised treatment. Study registration: This study is registered as PROSPERO CRD42018100572. 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. 73. See the NIHR Journals Library website for further project information.

Background Eating, drinking and swallowing difficulties (EDSD) are common in children with neurodisability, and have physical and non-physical causes. EDSD have substantial impacts on the child and family. Little is currently documented about what advice is usually given by professionals, including the interventions commonly used, and what informally constitutes ‘best clinical practice’. We aimed to identify current UK practice of parent-delivered interventions for EDSD for children with neurodisability, and the outcomes valued by professionals and parents.Methods Two populations were sampled: health professionals working with children and young people (aged 0–18 years) with neurodisability who experience EDSD (n=421); parents of children with neurodisability aged up to 12 years who experience EDSD (n=359). Questionnaires were developed based on the findings from updates of three systematic reviews, a mapping review of interventions used with this population, and in consultation with health professionals and parents. The questionnaires were distributed through UK health professional and parent networks and mainstream and specialist schools.Results Diverse professional groups, including speech and language therapists, occupational therapists, paediatricians and dietitians, support children with EDSD and neurodisability. A range of parent-delivered interventions, such as food and drink modification, positioning and modification of mealtime environment, were recommended by health professionals and are used by and acceptable to parents. Health professionals thought the interventions were effective but parents’ views were less consistent. Both health professionals and parents rated better general health and improved nutrition as the most important outcomes.Conclusions These survey findings outline current UK practice of parent-delivered interventions for EDSD in young children with neurodisability. The survey suggests key outcomes to measure in assessing the effectiveness of interventions. Further research is now needed to fully evaluate the effectiveness of interventions and move towards an evidence-based approach to best practice.

# Background Repetitive pitching places tremendous forces on the shoulder and elbow which can lead to upper extremity (UE) or lower extremity (LE) overuse injuries. # Purpose The purpose of this study was to evaluate pre-season physical measurements in collegiate baseball players and track in-season baseball throwing volume to determine which factors may predict throwing overuse injuries. # Study Design Retrospective Cohort study. # Methods Baseline preseason mobility, strength, endurance, and perception of function were measured in 17 collegiate baseball pitchers. Participants were then followed during the course of the season to collect rate of individual exposure, estimated pitch volume, and rating of perceived exertion in order to determine if changes in workload contributed to risk of injury using an Acute-to-Chronic Workload ratio (ACWR). # Results Participants developing an injury had greater shoulder internal rotator strength (p=0.04) and grip strength in a neutral position (p=0.03). A significant relationship was identified between ACWR and UE injuries (p

Abstract Dementia with Lewy bodies (DLB) is an age-associated neurodegenerative disorder producing progressive cognitive decline that interferes with normal life and daily activities. Neuropathologically, DLB is characterised by the accumulation of aggregated α-synuclein protein in Lewy bodies and Lewy neurites, similar to Parkinson’s disease (PD). Extrapyramidal motor features characteristic of PD, are common in DLB patients, but are not essential for the clinical diagnosis of DLB. Since many PD patients develop dementia as disease progresses, there has been controversy about the separation of DLB from PD dementia (PDD) and consensus reports have put forward guidelines to assist clinicians in the identification and management of both syndromes. Here, we present basic concepts and definitions, based on our current understanding, that should guide the community to address open questions that will, hopefully, lead us towards improved diagnosis and novel therapeutic strategies for DLB and other synucleinopathies.

Abstract Background Parents of babies admitted to neonatal units experience an arduous emotional journey. Feelings of helplessness, fear, sadness, guilt, grief and anger are common. These feelings can lead to anxiety, depression and post-traumatic stress which may persist long after discharge from the unit. Support from a parent with first-hand experience able to empathise with problems and challenges may help. This systematic review will identify quantitative and qualitative evidence to address the role of parent-to-parent support interventions for families of babies cared for in neonatal units, and combine the findings in an integrated synthesis. Methods We are working in collaboration with a study-specific Parent Advisory Group (PAG) of parents who have relevant and varied lived experience of having a baby in neonatal care and those who have been involved in providing peer support. With the PAG, we will carry out a systematic review bringing together all existing research on parent-to-parent support for parents of babies cared for in neonatal units. This will be reported in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. The protocol has been produced in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Protocol extension (PRISMA-P). We have co-produced a plain language protocol summary with the PAG which details the different stages of the project, and this is available via our website (http://clahrc-peninsula.nihr.ac.uk/research/parent-to-parent-support) for anyone interested in learning more about the detail of the project. Discussion All outputs will be available on the NIHR CLAHRC South West Peninsula (PenCLAHRC) website and promoted via PenCLAHRC networks as well as organisations that have been contacted throughout the project. PAG members will be involved in writing and reviewing the academic paper and final report and in co-producing dissemination products such as plain language summaries. The PAG will influence the main conclusions of the systematic review, aid interpretation and help to communicate results in the most appropriate ways. We will hold an impact conference with representatives from neonatal units, national neonatal networks, commissioners of services and parents to discuss what the findings mean for clinical practice and service provision. Systematic review registration PROSPERO CRD42018090569

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.

Abstract Background Parent carers of disabled children report poor physical health and mental wellbeing. They experience high levels of stress and barriers to engagement in health-related behaviours and with ‘standard’ preventive programmes (e.g. weight loss programmes). Interventions promoting strategies to improve health and wellbeing of parent carers are needed, tailored to their specific needs and circumstances. Methods We developed a group-based health promotion intervention for parent carers by following six steps of the established Intervention Mapping approach. Parent carers co-created the intervention programme and were involved in all stages of the development and testing. We conducted a study of the intervention with a group of parent carers to examine the feasibility and acceptability. Standardised questionnaires were used to assess health and wellbeing pre and post-intervention and at 2 month follow up. Participants provided feedback after each session and took part in a focus group after the end of the programme. Results The group-based Healthy Parent Carers programme was developed to improve health and wellbeing through engagement with eight achievable behaviours (CLANGERS – Connect, Learn, be Active, take Notice, Give, Eat well, Relax, Sleep), and by promoting empowerment and resilience. The manualised intervention was delivered by two peer facilitators to a group of seven parent carers. Feedback from participants and facilitators was strongly positive. The study was not powered or designed to test effectiveness but changes in measures of participants’ wellbeing and depression were in a positive direction both at the end of the intervention and 2 months later which suggest that there may be a potential to achieve benefit. Conclusions The Healthy Parent Carers programme appears feasible and acceptable. It was valued by, and was perceived to have benefited participants. The results will underpin future refinement of the intervention and plans for evaluation.

Abstract Background There is increasing recognition that establishing a core set of outcomes to be evaluated and reported in trials of interventions for particular conditions will improve the usefulness of health research. There is no established core outcome set for childhood epilepsy. The aim of this work is to select a core outcome set to be used in evaluative research of interventions for children with rolandic epilepsy, as an exemplar of common childhood epilepsy syndromes. Methods First we will identify what outcomes should be measured; then we will decide how to measure those outcomes. We will engage relevant UK charities and health professional societies as partners, and convene advisory panels for young people with epilepsy and parents of children with epilepsy. We will identify candidate outcomes from a search for trials of interventions for childhood epilepsy, statutory guidance and consultation with our advisory panels. Families, charities and health, education and neuropsychology professionals will be invited to participate in a Delphi survey following recommended practices in the development of core outcome sets. Participants will be able to recommend additional outcome domains. Over three rounds of Delphi survey participants will rate the importance of candidate outcome domains and state the rationale for their decisions. Over the three rounds we will seek consensus across and between families and health professionals on the more important outcomes. A face-to-face meeting will be convened to ratify the core outcome set. We will then review and recommend ways to measure the shortlisted outcomes using clinical assessment and/or patient-reported outcome measures. Discussion Our methodology is a proportionate and pragmatic approach to expediently produce a core outcome set for evaluative research of interventions aiming to improve the health of children with epilepsy. A number of decisions have to be made when designing a study to develop a core outcome set including defining the scope, choosing which stakeholders to engage, most effective ways to elicit their views, especially children and a potential role for qualitative research.

Effective science education is crucial for developing a scientifically literate citizenry, and for many, foundational primary science education experiences play a significant role in defining their long-term science trajectories. However, primary science education has been limited by a dissonance between the poor science trajectories for generations of primary students and the positive findings often reported university research; a divide that teachers are primarily responsible for bridging. This paper presents a scoping review of primary science intervention literature from the past 20 years to both describe the research outputs and analyse the evidence for the effectiveness of different primary science teaching approaches. The search yielded 142 research outputs from 26 nations with data from as many as 36,021 students. The results showed an established field with robust research designs covering all science disciplines and primary school years. Effect size analyses showed that an array of student-centred interventions covaried with large to very large improvements in science content knowledge, skills and attitudes. With the effectiveness of many student-centred approaches established in primary science education, issues of feasibility and scalability should now become a central focus for all stakeholders. Limited coverage of K-2 science in the sample was a point of concern.

Around the world, from the U.K. to India, governments and NGOs are formulating plans and raising funds to restore river and floodplain habitat. Much of this restoration work is undertaken in the interest of minimizing or rolling back the effects of disturbances, such as hurricanes, erosion, and urban development, and shoring up resilience, a river’s natural ability to resist disturbances. However, the words used to explain river systems have come to explain what threatens them, and to explain what river restoration must therefore accomplish. Words shape deeds.

Not so long ago, film music scholars complained habitually that, despite its established and powerful role in the media economy of film, music was being neglected by film and music studies alike. The multidisciplinary nature of film music studies was often cited as a possible reason for this state of affairs: film scholars felt they lacked sufficient musical education, or simply the terminology, to address the matter, while musicologists sniffed at what seemed to them a demeaning appropriation of music in the service of mere illustration or mood-setting. The division between film theory and film music theory was further exacerbated by the perception cultivated by some film scholars that music represented an addition to, rather than an integral part of, film (Kalinak). Not that the early literature on film music did much to suggest otherwise: largely anecdotal in nature, it documented the use of music in silent and early sound films in ways that seemed to reinforce the notion of music as post-production in practice and an after-thought to theory.

Background: Attention-deficit/hyperactivity disorder (ADHD) is a neurodevelopmental disorder characterised by age-inappropriate levels of inattention, impulsivity and hyperactivity. School can be particularly challenging for children with ADHD. Few reviews have considered non-pharmacological interventions in school settings. Objectives: To assess the effectiveness of non-pharmacological interventions delivered in school settings for pupils with, or at risk of, ADHD and to explore the factors that may enhance, or limit, their delivery. Data sources: Twenty electronic databases (including PsycINFO, MEDLINE, EMBASE, Education Resources Information Centre, The Cochrane Library and Education Research Complete) were searched from 1980 to February–August 2013. Three separate searches were conducted for four systematic reviews; they were supplemented with forward and backwards citation chasing, website searching, author recommendations and hand-searches of key journals. Review methods: The systematic reviews focused on (1) the effectiveness of school-based interventions for children with or at risk of ADHD; (2) quantitative research that explores attitudes towards school-based non-pharmacological interventions for pupils with ADHD; (3) qualitative research investigating the attitudes and experiences of children, teachers, parents and others using ADHD interventions in school settings; and (4) qualitative research exploring the experience of ADHD in school among pupils, their parents and teachers more generally. Methods of synthesis included a random-effects meta-analysis, meta-regression and narrative synthesis for review 1, narrative synthesis for review 2 and meta-ethnography and thematic analysis for reviews 3 and 4. Results: For review 1, 54 controlled trials met the inclusion criteria. For the 36 meta-analysed randomised controlled trials, beneficial effects (p

Background: The needs of children with autism spectrum disorder (ASD) are complex and this is reflected in the number and diversity of outcomes assessed and measurement tools used to collect evidence about children’s progress. Relevant outcomes include improvement in core ASD impairments, such as communication, social awareness, sensory sensitivities and repetitiveness; skills such as social functioning and play; participation outcomes such as social inclusion; and parent and family impact. Objectives: To examine the measurement properties of tools used to measure progress and outcomes in children with ASD up to the age of 6 years. To identify outcome areas regarded as important by people with ASD and parents. Methods: The MeASURe (Measurement in Autism Spectrum disorder Under Review) research collaboration included ASD experts and review methodologists. We undertook systematic review of tools used in ASD early intervention and observational studies from 1992 to 2013; systematic review, using the COSMIN checklist (Consensus-based Standards for the selection of health Measurement Instruments) of papers addressing the measurement properties of identified tools in children with ASD; and synthesis of evidence and gaps. The review design and process was informed throughout by consultation with stakeholders including parents, young people with ASD, clinicians and researchers. Results: The conceptual framework developed for the review was drawn from the International Classification of Functioning, Disability and Health, including the domains ‘Impairments’, ‘Activity Level Indicators’, ‘Participation’, and ‘Family Measures’. In review 1, 10,154 papers were sifted – 3091 by full text – and data extracted from 184; in total, 131 tools were identified, excluding observational coding, study-specific measures and those not in English. In review 2, 2665 papers were sifted and data concerning measurement properties of 57 (43%) tools were extracted from 128 papers. Evidence for the measurement properties of the reviewed tools was combined with information about their accessibility and presentation. Twelve tools were identified as having the strongest supporting evidence, the majority measuring autism characteristics and problem behaviour. The patchy evidence and limited scope of outcomes measured mean these tools do not constitute a ‘recommended battery’ for use. In particular, there is little evidence that the identified tools would be good at detecting change in intervention studies. The obvious gaps in available outcome measurement include well-being and participation outcomes for children, and family quality-of-life outcomes, domains particularly valued by our informants (young people with ASD and parents). Conclusions: This is the first systematic review of the quality and appropriateness of tools designed to monitor progress and outcomes of young children with ASD. Although it was not possible to recommend fully robust tools at this stage, the review consolidates what is known about the field and will act as a benchmark for future developments. With input from parents and other stakeholders, recommendations are made about priority targets for research. Future work: Priorities include development of a tool to measure child quality of life in ASD, and validation of a potential primary outcome tool for trials of early social communication intervention. Study registration: This study is registered as PROSPERO CRD42012002223. Funding: The National Institute for Health Research Health Technology Assessment programme.

Background: The identification of suitable outcome measures will improve the evaluation of integrated NHS care for the large number of children affected by neurodisability, and has the potential to encourage the provision of more appropriate and effective health care. This research sought to appraise the potential of patient-reported outcome measures (PROMs) for children and young people with neurodisability. Aim: This research aimed (i) to identify key outcomes of health care for children with neurodisability, beyond morbidity and mortality, from the perspectives of children, parents and professionals; (ii) to critically appraise existing generic multidimensional PROMs; and (iii) to examine whether or not the key outcomes might be measured by existing PROMs. We also sought agreement on a definition of neurodisability. Methods: Data were gathered in three main ways, (i) a systematic review identified eligible generic multidimensional PROMs and peer-reviewed studies evaluating psychometric performance using English-language questionnaires. Studies were appraised for methodological quality and psychometric performance was appraised using standard criteria. (ii) Focus groups and interviews with children and young people with neurodisability, and separately with parents, sought to identify important outcomes of NHS care, and their feedback on example PROM questionnaires. (iii) An online Delphi survey was conducted with a multidisciplinary sample of health professionals to seek agreement on appropriate NHS outcomes. In addition, we convened a consensus meeting with a small nominal group of young people, parents and professionals; the group sought agreement on a core set of important health outcomes. Results: From the systematic review, we identified 126 papers that reported eligible evidence regarding the psychometric performance of 25 PROMs. Evidence of psychometric robustness was more favourable for a small number of PROMs: KIDSCREEN (generic), DISABKIDS (chronic-generic) and Child Health Utility 9D (preference-based measure). The Pediatric Quality of Life Inventory and KINDL offer both self-report and a proxy report version for a range of age bands, but evidence of their psychometric performance was weaker. Evidence was lacking in one or more respects for all candidate PROMs, in both general populations and those with neurodisability. Proxy reporting was found generally to be poorly correlated with self-report. Focus groups and interviews included 54 children and young people, and 53 parents. The more important health outcomes were felt to be communication, emotional well-being, pain, mobility, independence/self-care, worry/mental health, social activities and sleep. In addition, parents of children with intellectual impairment identified behaviour, toileting and safety as important outcomes. Participants suggested problems with the face validity of example PROM questionnaires for measuring NHS care. In the Delphi survey, 276 clinicians from a wide range of professions contributed to at least one of four rounds. Professionals rated pain, hearing, seeing, sleep, toileting, mobility and communication as key goals for the NHS but also identified treating neurological symptoms as important. Professionals in the Delphi survey and parents working with the research team agreed a proposed definition for neurodisability. The consensus meeting confirmed overlap between the outcomes identified as important by young people, parents and professionals, but not complete agreement. Conclusions: There was agreement between young people, parents and professionals regarding a core suite of more important health outcomes: communication, emotional well-being, pain, mobility, independence/self-care, worry/mental health, social activities and sleep. In addition, behaviour, toileting and safety were identified as important by parents. This research suggests that it would be appropriate to measure these constructs using PROMs to assess health care. None of the candidate PROMs in the review adequately captures all of the identified constructs, and there is inadequate evidence that candidate PROMs are psychometrically robust for use across children with neurodisability. Further consultation with young people, families and professionals is warranted to support the use of PROMs to measure NHS outcomes. Research to test potential PROMs with different age groups and conditions would be valuable. Funding: The National Institute for Health Research Health Services and Delivery Research programme.