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Abstract Background In the UK, over 7,000 people are on the waiting list for an organ transplant and there are inequalities in need, access and waiting time for organs, with notable differences between minority ethnic groups. In May 2020, England changed the law and introduced a ‘soft’ opt-out system of consent to organ donation with a view to increase consent rates. We aimed to learn more about the impact of the law change on attitudes and views likely to be relevant to consent to deceased organ donation between different population subgroups. Methods Mixed-methods design involving latent class analysis of data from twelve repeated cross-sectional surveys undertaken from 2015 to 2021 (n = 19,011); analysis of the law change survey dataset collected quarterly from 2018 to 2022 (n = 45,439); and interviews with purposively selected members of the public (n = 30) with a focus on minority perspectives. Results Support for the principle of deceased organ donation remained high and stable in the general population (80%) but was 20% lower among ethnic minorities. From 2018 to 2022, an average of 58% of the general population was aware of the law change; this was lower among minority ethnic groups (31%). We identified four population subgroups (supportive donors (24% of the population); unengaged donors (22%); uncommitted donors (46%); and unsupportive donors (9%)). Interview themes included the challenges of discussing organ donation decisions, balancing autonomy with respecting family relationships, targeted misinformation, frustrations at the lack of consensus between community leaders, limited understanding of what happens during the end-of-life care leading to organ donation, and how this aligns with cultural values and preferences. Conclusion Implementation of the law change has not been associated to date with any change in public attitudes and preferences likely to influence consent overall or in minority ethnic groups in England. Uncommitted donors may benefit from encouragement to express their organ donation decision, and unengaged donors from attempts to address mis/information, confusion, and uncertainty. Interventions to raise the consent rate need to take account of the significant role of the family as well as wider community influences on attitudes, preferences and decision-making, particularly among certain minority (ethnic) groups.

Background Children and young people with experience of being in care (e.g. foster care, kinship care, residential care or at home with a supervision requirement order) are at higher risk of adverse mental health and well-being outcomes compared to the general population. Despite a range of policy recommendations and interventions, it is not clear what approaches are effective in the United Kingdom, or how context factors give rise to facilitators and inhibitors of implementation and acceptability. Objectives The CHIMES review is a complex-systems-informed mixed-method systematic review that aimed to synthesise the international evidence base for interventions addressing the mental health and well-being of care-experienced children and young people (age ≤ 25 years) and to assess the potential transportability of this evidence base to the United Kingdom context. Data sources We searched 16 electronic bibliographic databases and 22 websites from 1990 to May 2022. We conducted citation tracking, screened relevant systematic reviews and contacted international experts. Method We used a convergent synthesis design. We first constructed an evidence map to confirm review scope before undertaking method-level syntheses for outcome evaluations, process evaluations and economic evaluations. These elements were integrated into a review-level synthesis to identify potential evidence-based interventions that may progress to further development, adaptation and evaluation in the United Kingdom. We conducted stakeholder consultations to prioritise intervention theories, types and outcomes. Results We identified 64 interventions from 124 study reports. Interventions were primarily evaluated in the United States and targeted young people’s competencies or carers’ parenting practices. Meta-analysis reported limited evidence that interventions effectively improved mental health in the shorter term (0–6 months): total social, emotional and behavioural problems (d = −0.15, 95% confidence interval −0.28 to −0.02); internalising problem behaviours (d = −0.35, 95% confidence interval −0.61 to −0.08); externalising problem behaviours (d = −0.30, 95% confidence interval −0.53 to −0.08); depression and anxiety (d = −0.26, 95% confidence interval −0.40 to −0.13) and social–emotional functioning difficulties (d = −0.18, 95% confidence interval −0.31 to −0.05), but these impacts were not observed in the longer term (> 6 months). Five key context factors potentially explain challenges to implementation and acceptability: lack of system resources; the time, cognitive and emotional burden of delivery or participation; interprofessional tensions; the devaluing of young people, meaning that they felt unable to express dissatisfaction with interventions; and the devaluating of carers’ expertise and needs. From the evidence, stakeholder consultation identified two priority interventions: (1) mentoring by individuals with knowledge and experience of care and (2) system and ethos change to create harmonisation between organisations and facilitate interprofessional relationships. Well-being and suicide-related behaviours are priority outcomes alongside mental health. Limitations The review was limited by a paucity of theory and economic evaluations, so it is unclear how interventions might function or their potential cost-effectiveness. Interventions were insufficiently described, making it challenging to map the evidence base. Outcome evaluations were poorly reported. Due to ongoing restrictions with COVID-19, stakeholder consultations were conducted later than intended with a smaller number of attendees. Conclusions The review identified some evidence for interventions impacting mental health in the short term. There is a lack of system-level interventions and approaches that target subjective well-being and suicide-related outcomes. Future intervention might prioritise mentoring and targeting system culture. Study registration This study is registered as PROSPERO CRD42020177478. Funding This award was funded by the National Institute for Health and Care Research (NIHR) Public Health Research programme (NIHR award ref: NIHR129113) and is published in full in Public Health Research; Vol. 12, No. 14. See the NIHR Funding and Awards website for further award information. Plain language summary Children and young people in care may include those who live with foster carers, kinship carers or in residential care. Some care-experienced young people will have poorer mental health and well-being than those who have not been in care. In the United Kingdom, there have been policies and recommendations to improve mental health service provision, but it is not clear what works. We brought together all international research to understand what approaches work best and if they could be successfully used in the United Kingdom. We found 64 different types of mental health and well-being provision for children and young people up to the age of 25 years. Most approaches were delivered and evaluated in the United States. Generally, there was a lack of description of different provisions, but they often focused on improving young people’s social and emotional skills or changing the parenting style of foster carers. We found that, in the short term, these approaches could work to positively impact children and young people’s mental health, but in the longer term they did not seem to work. It is not clear if these approaches are more cost-effective than current services. We did not find much research looking at the impact on children and young people’s well-being, self-harm or suicide. We also considered some of the challenges to delivering services. Issues included a lack of time and finance, the burden on social care staff and carers, difficulty in relationships between organisations, services not meeting the needs of young people and carers struggling to deliver new approaches that do not fit with their skill set. After talking through our findings with stakeholders, we identified potential services that should be considered for delivery in the United Kingdom. These are mentoring for children and young people by individuals with experience of care and support to help carers, other professionals and organisations to work together. Scientific summary Background Care-experienced children and young people may be defined as individuals who have resided in kinship care, foster care, residential care or who remain at home but with statutory intervention that transfers legal parental rights to local authorities. They are reported to experience adverse mental health and well-being outcomes in comparison with the general population. Despite policy and guidance recommendations to improve the quality of support provided to care-experienced young people in the UK, the current evidence base for intervention in this context is limited. This is in contrast to a more comprehensive, if equivocal, evidence base internationally, particularly in the USA. There is a clear need for evidence syntheses that draw together evaluations reporting the effectiveness of different types of intervention approaches, while also exploring the contexts in which they are delivered and evaluated. Such work would help researchers and policymakers better understand the potential transportability of international evidence-based approaches beyond their immediate evaluation contexts, specifically to the UK. It is then important to establish the extent to which de novo intervention development, adaptation or revaluation is required for the UK setting. Objectives The Care-experienced cHildren and young people’s Interventions to improve Mental health and wEll-being outcomes Systematic review (CHIMES) review is a complex-systems informed, multimethod systematic review that aimed to synthesise extant international evidence on interventions addressing the mental health and well-being of care-experienced children and young people. This research aim was addressed through the following research questions (RQs): What are the types, theories and outcomes tested in mental health and well-being interventions for care-experienced children and young people? What are the effects (including inequities and harms) and economic effects of interventions? How do contextual characteristics shape implementation factors and what are key enablers and inhibitors of implementation? What is the acceptability of interventions to target populations? Can and how might intervention types, theories, components and outcomes be related in an overarching system-based programme theory? Drawing on the findings from RQ1 to RQ5, what do stakeholders think is the most feasible and acceptable intervention in the UK that could progress to further outcome or implementation evaluation? Methods We conducted a mixed-method systematic review, adopting a convergent synthesis design. This approach entailed method-specific syntheses conducted in a complementary manner, which were subsequently integrated into a further review-level synthesis. Data sources We searched 16 electronic bibliographic databases and 22 websites from 1990 to May 2022. A total of 32 subject experts and 17 third-sector organisations were contacted to identify additional grey literature, unpublished research or ongoing studies. We screened relevant systematic reviews identified at the protocol development stage and through the searches of electronic bibliographic databases. We conducted backward and forward citation tracking of included study reports. Data extraction We coded all eligible study reports as part of the review mapping, with intervention descriptions being coded using the Template for Intervention Description and Replication (TIDieR) checklist. Process evaluations were extracted according to context, implementation and acceptability. A subset of conceptually and/or empirically richer process evaluations were extracted according to the context and implementation of complex interventions framework, which classifies pertinent context domains. For outcome evaluations, randomised controlled trials (RCTs) and non-randomised studies were extracted according to study arms, analysis and outcomes, with study design-specific features also being coded. Equity harms were extracted from study reports that included moderator analysis or interaction effects. Harms were initially categorised according to the PROGRESS-Plus for equity harms. Economic evaluations coded according to the Drummond checklist. Quality appraisal We appraised programme theory study reports using a tailored appraisal tool developed for a previous systematic review with theory synthesis. Qualitative data within rich process evaluations were appraised using a tool developed in a previous systematic review, assessing reliability and trustworthiness. Outcome evaluations that were conducted using a RCT study design were appraised using the Cochrane risk of bias tool for randomised trials (RoB 2). Outcome evaluations that were conducted using a non-randomised study design were appraised using the Cochrane Risk Of Bias In Non-randomized Studies – of Interventions. For the assessment of certainty, we used Grading of Recommendations Assessment, Development and Evaluation (GRADE) and GRADE-Confidence in the Evidence from Reviews of Qualitative Research tools. Data synthesis Following the identification of eligible study reports, we constructed an evidence map to confirm the review scope and identify reports to be included in method-specific syntheses. Rich process evaluations were synthesised with framework synthesis. Thin-process evaluations, usually integrated with outcome evaluations, were descriptively summarised. For eligible RCT studies, we conducted meta-analyses for outcome domains relating to mental, behavioural or neurodevelopmental disorders as specified by the International Classification of Diseases, 11th Edition. There was not an adequate number of studies to conduct meta-analyses for the outcome domain of subjective well-being or suicide-related outcomes. We constructed narrative overviews for equity harms, with harvest plots for interventions targeting mental health, behavioural and neurodevelopmental disorders, as there was a sufficient number of study reports. Due to a lack of eligible economic evaluations, we narratively summarised one partial evaluation. We integrated the method-level syntheses into a review-level synthesis at two key points. First, we integrated the synthesis of thin and rich process evaluations (RQ3–4) with outcome data to explain intervention effectiveness and variations in effects (RQ2). Second, we constructed two integrative matrices. The first of these 2 × 2 matrices mapped interventions by stakeholder preferences (both in process evaluations and consultations) in regard to intervention theories and types. This was intended to identify whether the designs of interventions are relevant and responsive to needs within the UK context. The second mapped intervention outcomes by stakeholder priority outcomes to assess whether interventions are targeting the right domains. Stakeholder consultations At commencement of the review, we conducted stakeholder consultations with advisory groups of care-experienced young people and a foster carer manager advisory group to refine and confirm the review scope. Following completion of the method-level syntheses, we undertook seven stakeholder consultations with: two care-experienced young people’s advisory groups, one foster carer group, three health and social care practitioner groups and one government group. These consultations reflected on the evidence base and the potential transportability to the UK context. They considered whether identified intervention theories and types could be effective, feasible and acceptable in the UK, or if de novo developmental or adaptation would be required (RQ6). Results What are the types, theories and outcomes tested in mental health and well-being interventions for care-experienced children and young people? In total, 15,068 unique study reports were identified. Following screening, 64 interventions with 124 associated study reports were eligible for inclusion in the review. Study reports were published between 1994 and 2022, with the majority conducted solely in the USA (n = 77) or the USA and UK (n = 1). There were 24 study reports describing interventions’ programme theory, 50 process evaluations reporting context, implementation and acceptability, 86 outcome evaluations and 1 partial economic evaluation. We classified interventions according to the socioecological domains in which they operated, working on the assumption that they may interact with contextual characteristics differently depending on the part of the system they targeted. Of the interventions, 9 targeted the intrapersonal level, 15 targeted both the intrapersonal and interpersonal domain, 1 targeted the intrapersonal, organisational and community domains, 26 targeted the interpersonal domain, targeted the interpersonal and organisational domain, 5 targeted the interpersonal, organisational and community domain, 1 targeted the organisational domain, 4 targeted the community domain and 1 targeted the policy domain. The 13 interventions reporting a programme theory were mainly relational and focused on attachment theory, positive youth development and social learning theory. This reflected the predominance of interpersonal interventions in the review. There was also system change theories linked to interventions operating at the higher socioecological domains, although these generally focused on restructuring the system to support interpersonal approaches. Interventions primarily targeted mental health, behavioural and neurodevelopmental disorders. The most frequently assessed outcome measurements were total social, emotional and behavioural problems (n = 48); social–emotional functioning difficulties (n = 17); externalising problem behaviours (n = 26) and internalising problem behaviours (n = 22). Only 11 interventions targeted subjective well-being and 4 targeted suicide-related behaviours. What are the effects (including inequities and harms) and economic effects of interventions? We synthesised evidence from 44 RCT evaluations of 35 interventions. Meta-analyses showed that interventions reporting outcomes for up to 6 months post baseline demonstrated some effectiveness for reducing children and young people’s: total social, emotional and behavioural problems [d = −0.15, 95% confidence interval (CI) −0.28 to −0.02]; internalising problem behaviours (d = −0.35, 95% CI −0.61 to −0.08); externalising problem behaviours (d = −0.30, 95% CI −0.53 to −0.08); depression and anxiety (d = −0.26, 95% CI −0.40 to −0.13) and social–emotional functioning difficulties (d = −0.18, 95% CI −0.31 to −0.05). Assessment of evidence using GRADE showed low or very low certainty across outcome domains, primarily relating to concerns arising from risk of bias and imprecision across evaluation reports. For outcome domains where there were a sufficient number of effect sizes to evaluate longer-term (> 6 months) outcomes (total social, emotional and behavioural problems; internalising problem behaviours; externalising problem behaviours and social–emotional functioning difficulties), we found no evidence that interventions demonstrated effectiveness. Evidence of equity harms indicated limited differential outcomes according to population groups. However, there was some tentative indication that interventions targeting mental health, behavioural and neurodevelopmental disorders were more beneficial for those with less exposure to maltreatment and those with more severe baseline mental health problems. The review only identified one partial evaluation of an included intervention assessing intervention costs in the UK relative to the USA. How do contextual characteristics shape implementation factors, and what are key enablers and inhibitors of implementation? What is the acceptability of interventions to target populations? We categorised process evaluations according to conceptually and/or empirically thin (n = 27) or rich (n = 23). Generally, thin-process evaluations indicated that interventions had high fidelity and acceptability, although there were reported issues with recruitment and retention. From rich-process evaluations, we generated five key context themes that might serve as facilitators or inhibitors to implementation and acceptability: (1) lack of system resources; (2) the time, cognitive and emotional burden of delivery and participation; (3) tensions in interprofessional relationships; (4) the systemic devaluing of care-experienced young people where their needs and preferences are not prioritised and (5) the discounting of carers’ expertise, knowledge and other potentially conflicting commitments, which can mean that interventions do not fit with the wider context of their lives. There was no clear difference between interventions that reported high levels of implementation and/or acceptability and effectiveness. Can and how might intervention types, theories, components and outcomes be related in an overarching system-based programme theory? From our mapping and synthesis of theory, outcome and process evaluations, we identified three clusters of interventions that might have potential to progress to further testing in the UK. Within these clusters of intervention types, there were specific programme theories or components that demonstrated some evidence of effectiveness. Two of these approaches primarily operate at the interpersonal level: (1) mentoring interventions delivered by care-experienced peers or significant adults with knowledge or experience of care and (2) parenting interventions, largely targeted at foster and kinship carers, that provide training and support in parenting skills, knowledge and practices. The third type targets the organisational and community domains, and comprises system-change interventions facilitating interorganisational relationships and collaboration, largely through the harmonisation of ethos. Currently, these types of interventions primarily target mental health, behavioural and neurodevelopmental disorders, although there are examples of each intervention type addressing subjective well-being and self-harm. From the evidence base, these approaches have not been combined into an overarching intervention model, although they are not theoretically discordant and might have the potential for integration. Drawing on the findings from research questions 1–5, what do stakeholders think is the most feasible and acceptable intervention in the United Kingdom that could progress to further outcome or implementation evaluation? Reflecting on the evidence synthesis, consultations with stakeholders refined key context factors, in addition to identifying priority intervention theories, types and outcomes that can inform further intervention development, adaptation and evaluation in the UK. Stakeholders confirmed the key context factors generated by the process evaluation synthesis as being relevant facilitators or inhibitors to intervention implementation and acceptability in the UK. Priority intervention types emphasised mentoring approaches, preferably by care-experienced peers, and system change approaches where harmonisation in ethos across professional groups and community organisations works to facilitate interagency working in decision-making and service co-ordination. These intervention types may be underpinned by theories that have an emphasis on positive relationships (e.g. attachment theory, positive youth development, and social learning theory) and progress understanding of the particular challenges and complexities experienced by young people in care (e.g. trauma-informed practice). Parenting interventions were not considered a priority where they were theoretically aligned with behavioural management. Priority outcomes for stakeholders were subjective well-being and suicide-related behaviours. Interventions may be adapted to also assess these outcomes where theoretically appropriate or de novo development may be required. Conclusions The available evidence base reporting on interventions targeting the mental health and well-being of care-experienced children and young people is mixed, and is limited for certain intervention theories, types and outcomes. The evidence base, primarily from the USA, focuses on intrapersonal and interpersonal approaches that develop the skills and knowledge of young people and their carers. Current interventions primarily target mental, behavioural and neurodevelopmental disorders. We identified mentoring and system ethos change interventions as being a priority in the UK context, provided additional developmental and adaptation work is undertaken to sensitise these types of approaches to local system needs. Study registration This study is registered as PROSPERO CRD42020177478. Funding This award was funded by the National Institute for Health and Care Research (NIHR) Public Health Research programme (NIHR award ref: NIHR129113) and is published in full in Public Health Research; Vol. 12, No. 14. See the NIHR Funding and Awards website for further award information.

Background Childhood chronic pain is a widespread public health issue. We need to understand how children with chronic pain and their families experience chronic pain and its management. Objectives To conduct a meta-ethnography on the experiences and perceptions of children with chronic pain and their families of chronic pain, treatments and services. We investigated how children and their families conceptualise and live with chronic pain; what they think of and want from health and social care services; and what they conceptualise as ‘good’ pain management. Design Meta-ethnography with stakeholder and patient and public involvement in the design, search and sampling strategies, analysis and dissemination. Review strategy: comprehensive searches of 12 bibliographic databases and supplementary searches in September 2022, to identify qualitative studies with children aged 3 months to 18 years with chronic non-cancer pain and their families. We included studies with rich explanatory data; appraised methodological limitations using the Critical Appraisal Skills Programme tool; and extracted, analysed and synthesised studies’ findings. We used Grading of Recommendations Assessment, Development and Evaluation-confidence in the evidence from reviews of qualitative research to assess confidence in review findings. We integrated findings with 14 Cochrane treatment effectiveness reviews on children’s chronic non-cancer pain. Results We synthesised 43 studies sampled from 170 eligible studies reported in 182 publications. Studies had minor (n = 24) or moderate (n = 19) methodological limitations. Grading of Recommendations Assessment, Development and Evaluation-confidence in the evidence from reviews of qualitative research assessments of review findings were high (n = 22), moderate (n = 13) or very low confidence (n = 1). Moderate and severe chronic pain had profound adverse impacts on family members’ well-being, autonomy and self-identity; family dynamics; parenting approaches; friendships and socialising; children’s education and parental paid employment. Most children and families sought a biomedical cure for pain. They experienced difficulties seeking and receiving support from health services to manage pain and its impacts. Consequently, some families repeatedly visited health services. Cochrane reviews of intervention effects and trials did not measure some outcomes important to children and families, for example effects of pain on the family and resolution of pain. Reviews have mainly neglected a biopsychosocial approach when considering how interventions work. Limitations There were limited data on common pain conditions like migraine/headache, abdominal pain; some rarer conditions; children with learning disabilities and under-fives; siblings; fathers and experiences of treatments/services. We excluded studies on cancer, end-of-life pain and experiences of healthcare professionals. Conclusions We developed the family-centred theory of children’s chronic pain management, integrating health and social care with community support. Future work Future research should explore families’ experiences of services and treatments, including opioids, and social care services; experiences of children with autism and learning disabilities, under 5 years old and with certain common pain conditions. We need development and testing of family-centred interventions and services. Study registration This study is registered as PROSPERO (CRD42019161455) and Cochrane Pain, Palliative and Supportive Care (623). Funding This award was funded by the National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research programme (NIHR award ref: NIHR128671) and is published in full in Health and Social Care Delivery Research; Vol. 12, No. 17. See the NIHR Funding and Awards website for further award information. Plain language summary Chronic pain lasting 3 months or more affects at least 8% of children in the UK. It causes difficulties with many aspects of children’s lives including relationships, school attendance and use of National Health Services. In the UK, there are few specialist services and a lack of high-quality research for managing children’s chronic pain. Our study aimed to locate and pull together existing research on the experiences and views of children with chronic pain and their families. We wanted to find out how they think about and live with chronic pain, and their views and experiences of services and treatments, and what they want to achieve from them. We conducted this study alongside children with chronic pain and their families, charities, healthcare professionals and academic experts. They helped us to conduct the study and to ensure our findings are relevant to children, families and the National Health Service. We pulled together data from 43 studies that best answered our questions. We found that moderate and severe children’s chronic pain that was not well managed affected the whole family, including their relationships and social lives. Families found it difficult to get help and a diagnosis from health services. Most families wanted a medical cure for pain. Families had long waits for answers and treatment but gradually realised there may be no cure, so they focused on living well with pain or gave up hope. Children and families from ethnic minority groups or with a learning disability experienced discrimination. Few studies focused on children under 5 years old, children with learning disabilities or experiences of services. Families need a pain management approach tailored to the whole family’s needs involving schools, social care and health services. Our findings could improve treatment guidelines, training of health and social care professionals and service design and treatments. Scientific summary Background Chronic pain in childhood is widespread, affecting at least 8% of children and young people (CYP) in the UK. For a sizeable number of children, chronic non-cancer pain has considerable negative impacts on children’s lives and quality of life and leads to increased use of healthcare services and medication. Treating adolescent pain alone has an annual cost of around 4 billion pounds in the UK. Provision of, and access to, services for managing children’s chronic pain is limited. We also do not know which treatment outcomes children and families value. To design and deliver services and treatments, which meet the needs of patients and their families, it is vital that we understand how children with chronic non-cancer pain and their families experience chronic pain, pain treatments and services, and which outcomes are important to them. Qualitative research addressing these urgent, important issues exists but must be synthesised to interpret the body of evidence. Existing qualitative evidence syntheses have focused on either a narrow chronic pain population or topic and none produced a theory. Therefore, we conducted a qualitative evidence synthesis using meta-ethnography, which is ideal for synthesising the diverse contexts of children’s chronic pain research and developing theory. We refer to the meta-ethnography by the acronym ‘CHAMPION’ (Children And young people’s Meta-ethnography on Pain). The meta-ethnography was conceived as a Cochrane review to inform and supplement existing Cochrane reviews of intervention effectiveness for children’s chronic pain. Cochrane requires qualitative evidence syntheses to be integrated with intervention effects reviews to further understanding of patient experiences, acceptability and implementation of interventions, which outcomes are important to children and their families, and inform further intervention development. Aim To conduct a meta-ethnography on the experiences and perceptions of CYP with chronic pain, and their families, of chronic pain, treatments and services to inform the design and delivery of health and social care services, interventions and future research. Review questions How do children with chronic pain and their families conceptualise chronic pain? How do they live with chronic pain? What do they think of how health and social care services respond to and manage their/their child’s chronic pain? What do they conceptualise as ‘good’ chronic pain management and what do they want to achieve from chronic pain management interventions and services? Objectives Conduct comprehensive searches to identify qualitative research literature on the experiences and perceptions of children with chronic pain and their families to address review questions 1–4. Select and synthesise relevant studies using meta-ethnography. Ensure salience of findings via involvement of children with chronic pain and their families in study design, analysis and interpretation. Assess how much confidence can be placed in our synthesised findings using GRADE-CERQual (Grading of Recommendations Assessment, Development and Evaluation-confidence in the evidence from reviews of qualitative research) in order to facilitate use of our findings for NHS decision-making. Identify research gaps regarding review questions 1–4 in order to inform future research directions. Integrate our findings with existing relevant Cochrane treatment effectiveness reviews in order to determine if programme theories and outcomes of interventions match children and their families’ views. Inform the selection and design of patient-reported outcome measures for use in chronic pain studies and interventions and care provision to children and their families. Disseminate findings to academic, clinical, lay and policy audiences to influence childhood chronic pain policy and practice. Methods Design We conducted a meta-ethnography, a seven-phase interpretive methodology, which takes into account the contexts and meanings of the original studies. Patient and public involvement Patient and public involvement (PPI) of children with chronic non-cancer pain and their families was fundamental to all aspects of our meta-ethnography conduct from study inception to dissemination. We also worked with a Project Advisory Group of wider stakeholders. The groups approved the aim and design and search strategy. They participated in workshops to decide study inclusion and sampling criteria decisions, how to group studies for analysis, data interpretation, identification of research gaps and to develop outputs. They also informed and assisted with dissemination of findings. Review strategy We carried out comprehensive searches of 12 bibliographic databases including MEDLINE (medical literature analysis and retrieval system online), CINAHL (Cumulative Index to Nursing and Allied Health Literature), PsycInfo and 9 others, with iterative supplementary searches, including citation tracking. We searched for studies that had been published up to September 2022. Selection criteria To identify qualitative research studies with children aged 3 months to 18 years with chronic non-cancer pain, and their families, which focused on their perceptions, experiences and views of chronic pain, services and treatments. The final inclusion criteria were agreed with our PPI group. We included qualitative research studies of any design using recognisable qualitative methods of data collection, in any language, with no date restrictions. We did not include children with chronic pain related to cancer or end of life because they have distinct care pathways; Cochrane reviews of effects also synthesise non-cancer pain separately. Data collection and analysis Eligible studies were purposively sampled to ensure we could conduct an in-depth, synthesis of the data most relevant to answering our review questions. Sampling, organising studies for synthesis and analysis and interpretation involved our PPI group via two workshops and additional communication by teleconference, e-mail and social media. ‘Conceptually rich’ and ‘thick’ studies, that is, those with in-depth explanatory/interpretive findings and contextual details, were included. Sampling was guided by Cochrane Qualitative and Implementation Methods Group Guidance, and our advisory and involvement groups who agreed we should include non-UK studies and studies with moderately rich data to ensure a wider range of pain conditions were represented. We assessed methodological limitations of studies using the Critical Appraisal Skills Programme tool. We extracted data on study aims, focus and characteristics into Microsoft Excel® and findings using NVivo software. We compared these study data to determine how studies related to one another and decided how to order and group them for synthesis. Stakeholders and our PPI group agreed we should group studies by pain condition. We used meta-ethnography to synthesise each group of studies separately before synthesising them all together. Analysis and interpretation of studies involved children with chronic non-cancer pain and their families and has resulted in theory to inform service design and delivery. Our PPI group clarified the meaning of ambiguous study findings and identified and addressed gaps in the data. We used the GRADE-CERQual approach to assess our confidence in each key review finding and used a matrix approach to integrate our findings with 14 existing Cochrane reviews on treatment effectiveness for children’s chronic non-cancer pain. Results One hundred and seventy studies met inclusion criteria. We sampled and synthesised 43 most relevant conceptually rich and thick studies, which had minor (n = 24) or moderate (n = 19) methodological limitations overall. Using GRADE-CERQual, we rated 36 descriptive-level review findings as high (n = 22) or moderate (n = 13) confidence with only one very low confidence. Twenty-three included studies were conducted in the UK, 17 in other high-income countries and 4 in low- to middle-income countries (there is a three-country study, so the total does not add to 43 studies). Included studies involved 325 CYP with chronic pain, most aged 10 years or older, and 308 family members, mostly mothers. The studies covered chronic pain conditions including complex regional pain syndrome, juvenile idiopathic arthritis, sickle cell disease, headache, migraine, abdominal pain, musculoskeletal pain, epidermolysis bullosa, dysmenorrhoea, neurological conditions such as cerebral palsy, unspecified chronic pain or a mix of conditions. Moderate and severe children’s chronic pain had profound adverse impacts on the whole family, family life and their wider social connections. Pain affected all aspects of an individual’s and a family’s functioning: family dynamics and relationships; family members’ emotions, well-being, autonomy and a sense of self-identity; parenting strategies; friendships and socialising; children’s education and future employment prospects; and parental employment. Families experienced difficulties in seeking and getting help and support from health services to manage their child’s pain and its impacts. Most pain management occurred outside of health services supervised by parents, especially mothers who were usually the main caregivers. Children and young people and their families were aware of the biopsychosocial (physical/biological, psychological and social) impacts of chronic pain on their lives. However, most CYP and parents understood the cause of chronic pain as having solely an underlying biological cause. This influenced their expectations of, and interactions with, health services. However, families’ conceptualisations of pain were dynamic and could change over time influenced by experiences of treatments, services and interactions with healthcare professionals. Interactions with healthcare professionals were frequently negative and stressful. CYP and parents, particularly mothers, felt they were not listened to and believed, and thus felt unsupported. For instance, CYP’s experiences of living with pain were ignored or their pain was disbelieved, and mothers felt blamed for their child’s pain. Parents of younger children and of children with communication difficulties, in particular, perceived that healthcare professionals did not always recognise their expertise in assessing their child’s pain. A key challenge for children and their families was that they found health services fragmented and very difficult to navigate. Consequently, it was difficult to achieve satisfactory pain management. Families often entered a lengthy cycle, which could last many years, of attending then reattending health services, and awaiting referral. In addition to these navigation challenges, CYP and their families, particularly CYP with sickle cell disease and those with chronic pain and learning disabilities, such as Down syndrome, experienced prejudice and discrimination in health services. We created a model of how families navigate chronic management with two key routes labelled Path A and B. On Path A, families seek a cure from services but often repeatedly seek services without pain resolution. When their expectations of services are not met, they try to deal with pain alone, often unsuccessfully, which can dominate family life. Psychological treatments are only used as a last option. Path B depicts effective communication and expectation management by healthcare professionals which can help families to shift their focus from a cure to trying to live well despite the pain. Families might also realise independently over time that a cure is unlikely. Families on Path B tend to be more receptive to biopsychosocial approaches for pain management, due to lack of other options. Families can move between the paths depending on input from services and if pain worsens. We developed new interpretive findings resulting in the family-centred theory of children’s chronic pain and its management, which incorporates how children and families conceptualise pain, their experiences of living with pain and of pain management services, and defines ‘good’ pain management and services. Our mid-range theory describes good pain management and services as holistic, biopsychosocial, family-centred pain management which acknowledges the impact of CYP’s chronic pain on all aspects of family life and their social relationships. In the theory, pain management strategies are tailored to the family’s needs. Schools, social services, social care and healthcare services function as inter-related aspects in a whole system approach. This approach aims to reduce service fragmentation making services more intuitive for families. Empathetic communication is a core component of this approach, and it is fundamental for building trust between families and services. The theory includes assessment of chronic pain and of families’ psychosocial needs to enable effective triage and trigger appropriate actions from/referrals to different services (schools, social care and health services). Cochrane systematic reviews of intervention effects and trials did not measure some outcomes important to children and families, including effects of chronic pain on the family and resolution of pain. Reviews have mainly neglected a biopsychosocial approach when considering how interventions work. Limitations There were few conceptually rich qualitative studies focusing on common pain conditions like migraine, headache, abdominal pain; some rarer conditions; children with learning disabilities and under 5 years old; siblings; boys with chronic pain; fathers; and experiences of treatments/services for chronic pain. Our findings may not well represent the experiences and perceptions of the poorly represented groups and there were gaps in the evidence regarding experiences and perceptions of treatments/services. Three per cent (n = 1) of our findings were assessed as very low confidence, none were low confidence and 36% (n = 13) were moderate confidence, using GRADE-CERQual. Additional evidence could increase our confidence in those findings. We excluded studies focusing on chronic cancer pain, end-of-life pain management and experiences of healthcare professionals. Conclusions Currently, children’s chronic pain management does not involve holistic, biopsychosocial health and social care tailored for children and integrated with support in the community, which considers the needs of the entire family and the impact on the family life. The theory and findings developed in this meta-ethnography have the potential to contribute to the development and updating of existing clinical guidelines on children’s chronic non-cancer pain; inform training of health and social care professionals regarding children’s chronic pain management; and inform service, treatment and intervention design and delivery. Ultimately, this could improve the care provided and thus the health and quality of life of children with chronic non-cancer pain and their families, which in turn could reduce the burden on health and social care services. Findings are being disseminated widely to academic, lay, clinical and policy audiences. Outputs include a short, animated film on YouTube for children (https://youtu.be/Oz5I7xKYf5o), academic journal articles, a webinar, a lay podcast, an infographic, a policy briefing and other social media outputs. In addition, we have delivered eight conference presentations. Future work Family-centred outcomes should be included in future trials of chronic pain interventions. Conceptually rich, primary qualitative studies are needed to explore families’ experiences of services and treatments, including pain management plans, pain assessment, opioid use in children and social care services; experiences of children with autism and learning disabilities, under 5 years old, and with certain common pain conditions, such as headache, migraine and abdominal pain and with some rarer conditions such as Ehlers–Danlos syndrome; experiences of siblings and fathers of children with chronic pain; and healthcare professionals’ experiences and perceptions of chronic pain management. A qualitative evidence synthesis on chronic pain management for children with cancer is needed. Development and testing of family-centred children’s chronic pain interventions, services and treatments are needed. Study registration This study is registered as PROSPERO, the International Prospective Register of Systematic Reviews (reference: CRD42019161455) and Cochrane Pain, Palliative and Supportive Care (review number 623). Funding This award was funded by the National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research programme (NIHR award ref: NIHR128671) and is published in full in Health and Social Care Delivery Research; Vol. 12, No. 17. See the NIHR Funding and Awards website for further award information.

England switched to an opt-out system of consent in 2020 aiming to increase the number of organs available. Spain also operates an opt-out system yet has almost twice the organ donations per million population compared with England. We aimed to identify both differences and similarities in the consent policies, documents and procedures in deceased donation between the two countries using comparative qualitative content and discourse analysis. Spain had simpler, locally tailored documents, the time taken for families to review and process information may be shorter, there were more pathways leading to organ donation in Spain, and more robust legal protections for the decisions individuals made in life. The language in the Spanish documents was one of support and reassurance. Documents in England by comparison appeared confusing, since additions were designed to protect the NHS against risk and made to previous document versions to reflect the law change rather than being entirely recast. If England’s ambition is to achieve consent rates similar to Spain this analysis has highlighted opportunities that could strengthen the English system-by giving individuals’ decisions recorded on the organ donor register legal weight, alongside unifying and simplifying consent policies and procedures to support families and healthcare professionals.

Abstract Background Provision of and access to paediatric end-of-life care is inequitable, but previous research on this area has focused on perspectives of health professionals in specific settings or children with specific conditions. This qualitative study aimed to explore regional perspectives of the successes, and challenges to the equitable coordination and delivery of end-of-life care for children in the UK. The study provides an overarching perspective on the challenges of delivering and coordinating end-of-life care for children in the UK, and the impact of these on health professionals and organisations. Previous research has not highlighted the successes in the sector, such as the formal and informal coordination of care between different services and sectors. Methods Semi-structured interviews with Chairs of the regional Palliative Care Networks across the UK. Chairs or co-Chairs (n = 19) of 15/16 Networks were interviewed between October-December 2021. Data were analysed using thematic analysis. Results Three main themes were identified: one standalone theme (“Communication during end-of-life care”); and two overarching themes (“Getting end-of-life services and staff in the right place”, with two themes: “Access to, and staffing of end-of-life care” and “Inconsistent and insufficient funding for end-of-life care services”; and “Linking up healthcare provision”, with three sub-themes: “Coordination successes”, “Role of the networks”, and “Coordination challenges”). Good end-of-life care was facilitated through collaborative and network approaches to service provision, and effective communication with families. The implementation of 24/7 advice lines and the formalisation of joint-working arrangements were highlighted as a way to address the current challenges in the specialism. Conclusions Findings demonstrate how informal and formal relationships between organisations and individuals, enabled early communication with families, and collaborative working with specialist services. Formalising these could increase knowledge and awareness of end of life care, improve staff confidence, and overall improve professionals’ experiences of delivering care, and families’ experiences of receiving it. There are considerable positives that come from collaborative working between different organisations and sectors, and care could be improved if these approaches are funded and formalised. There needs to be consistent funding for paediatric palliative care and there is a clear need for education and training to improve staff knowledge and confidence.

Abstract Background GRADE-CERQual (Confidence in the Evidence from Reviews of Qualitative Research) is a methodological approach to systematically and transparently assess how much confidence decision makers can place in individual review findings from qualitative evidence syntheses. The number of reviews applying GRADE-CERQual is rapidly expanding in guideline and other decision-making contexts. The objectives of this evaluation were, firstly, to describe the uptake of GRADE-CERQual in qualitative evidence synthesis by review authors and, secondly, to assess both reporting of and fidelity to the approach. Methods The evaluation had two parts. Part 1 was a citation analysis and descriptive overview of the literature citing GRADE-CERQual. Authors worked together to code and chart the citations, first by title and abstract and second by full text. Part 2 was an assessment and analysis of fidelity to, and reporting of, the GRADE-CERQual approach in included reviews. We developed fidelity and reporting questions and answers based on the most recent guidance for GRADE-CERQual and then used NVivo12 to document assessments in a spreadsheet and code full-text PDF articles for any concerns that had been identified. Our assessments were exported to Excel and we applied count formulae to explore patterns in the data. We employed a qualitative content analysis approach in NVivo12 to sub-coding all the data illustrating concerns for each reporting and fidelity criteria. Results 233 studies have applied the GRADE-CERQual approach, with most (n = 225, 96.5%) in the field of health research. Many studies (n = 97/233, 41.6%) were excluded from full fidelity and reporting assessment because they demonstrated a serious misapplication of GRADE-CERQual, for example interpreting it as a quality appraisal tool for primary studies or reviews. For the remaining studies that applied GRADE-CERQual to assess confidence in review findings, the main areas of reporting concern involved terminology, labelling and completeness. Fidelity concerns were identified in more than half of all studies assessed. Conclusions GRADE-CERQual is being used widely within qualitative evidence syntheses and there are common reporting and fidelity issues. Most of these are avoidable and we highlight these as gaps in knowledge and guidance for applying the GRADE-CERQual approach.

Abstract Background The mental health and wellbeing of care-experienced children and young people (i.e. foster care, kinship care, residential care) is poorer than non-care-experienced populations. The Care-experienced cHildren and young people’s Interventions to improve Mental health and wEll-being outcomes Systematic review (CHIMES) aimed to synthesise the international evidence base for interventions targeting subjective wellbeing, mental health and suicide amongst care-experienced young people aged ≤ 25 years. Methods For the first phase of the review, we constructed an evidence map identifying key clusters and gaps in interventions and evaluations. Studies were identified through 16 electronic databases and 22 health and social care websites, in addition to expert recommendations, citation tracking and screening of relevant systematic reviews. We charted interventions and evaluations with a summary narrative, tables and infographics. Results In total, 64 interventions with 124 associated study reports were eligible. The majority of study reports were from the USA (n = 77). Interventions primarily targeted children and young people’s skills and competencies (n = 9 interventions), the parental functioning and practices of carers (n = 26), or a combination of the two (n = 15). While theoretically under-specified, interventions were largely informed by theories of Attachment, Positive Youth Development, and Social Learning Theory. Current evaluations prioritised outcomes (n = 86) and processes (n = 50), with a paucity of study reports including theoretical descriptions (n = 24) or economic evaluations (n = 1). Interventions most frequently targeted outcomes related to mental, behavioural or neurodevelopmental disorders, notably total social, emotional and behavioural problems (n = 48 interventions) and externalising problem behaviours (n = 26). There were a limited number of interventions targeting subjective wellbeing or suicide-related outcomes. Conclusions Future intervention development might focus on structural-level intervention theories and components, and target outcomes related to subjective wellbeing and suicide. In accordance with current methodological guidance for intervention development and evaluation, research needs to integrate theoretical, outcome, process and economic evaluation in order to strengthen the evidence base. Systematic review registration PROSPERO CRD42020177478.

Background: Globally, the demand for hospice care continues to grow resulting in substantial resource burden. Whilst some countries are able to rely on fixed government contributions, statutory funding for palliative care in the United Kingdom is unequally distributed. These unstable funding streams and increased demand means that hospices need to evidence their value. Objective: This study explored the experiences of patients and family-caregivers to determine what they valued most from accessing hospice services in Wales. Methods: In this large multi-site qualitative study, 94 semi-structured interviews and 2 focus groups were conducted with hospice patients ( n = 45), family-caregivers ( n = 18), hospice staff ( n = 31) and volunteers ( n = 10). The audio recordings were transcribed verbatim and analysed using Framework analysis. Results: Seven themes described patient and family-caregiver experiences and what they valued most: relationships with staff and volunteers, greater support networks which reduced social isolation and loneliness, provision of information and advice which improved patient autonomy, symptom management and subsequent reduction in psychological distress, improvements in patient functionality, mobility and overall physical health and respite relief which promoted improved relationships. Conclusion: This is the largest study to explore what patients and family-caregivers value from hospice care. Findings indicate that hospice care provides a truly needs-led and strengths-based service to those who are nearing and at the end-of-life, which is highly valued by patients and family members.

Objectives To identify the factors that determine treatment choices following pre-dialysis education.Design Retrospective cohort study using data linkage with univariate and multivariate analyses using linked data.Setting Secondary care National Health Service Wales healthcare system.Participants All people in Wales over 18 years diagnosed with established kidney disease, who received pre-dialysis education between 1 January 2016 and 12 December 2018.Main outcome measures Patient choice of dialysis modality and any kidney replacement therapy started.Results Mean age was 67 years; n=1207 (60%) were male, n=878 (53%) had ≥3 comorbidities, n=805 (66%) had mobility problems, n=700 (57%) had pain symptoms, n=641 (52%) had anxiety or were depressed, n=1052 (61.6%) lived less than 30 min from their treatment centre, n=619 (50%) were on a spectrum of frail to extremely vulnerable. n=424 (25%) chose home dialysis, n=552 (32%) chose hospital-based dialysis, n=109 (6%) chose transplantation, n=231 (14%) chose maximum conservative management and n=391 (23%) were ‘undecided’. Main reasons for not choosing home dialysis were lack of motivation/low confidence in capacity to self-administer treatment, lack of home support and unsuitable housing. Patients who choose home dialysis were younger, had lower comorbidities, lower frailty and higher quality of life scores. Multivariate analysis found that age and frailty were predictors of choice, but we did not find any other demographic associations. Of patients who initially chose home dialysis, only n=150 (54%) started on home dialysis.Conclusion There is room for improvement in current pre-dialysis treatment pathways. Many patients remain undecided about dialysis choice, and others who may have chosen home dialysis are still likely to start on unit haemodialysis.

BackgroundIn May 2020, England implemented soft 'opt-out' or 'deemed consent' for deceased donation with the intention of raising consent rates. However, this coincided with the COVID-19 pandemic, making it difficult to assess the early impact of the law change. Wales and Scotland changed their organ donation legislation to implement soft opt-out systems in 2015 and 2021 respectively. This study provides a descriptive analysis of changes in consent and transplant rates for deceased organ donation in England, Scotland and Wales.MethodsLogistic regression and descriptive trend analysis were employed to assess the probability of a patient who died in critical care becoming a donor, and to report consent rates using data, respectively, from the Intensive Care National Audit and Research Centre (ICNARC) in England from 1 April 2014 to 30 September 2021, and from the Potential Donor Audit for England, Scotland and Wales from April 2010 to June 2023.ResultsThe number of eligible donors in April-June 2020 were 56.5%, 59.3% and 57.6% lower in England, Scotland and Wales relative to April-June 2019 (pre-pandemic). By April-June 2023, the number of eligible donors had recovered to 87.4%, 64.2% and 110.3%, respectively, of their levels in 2019. The consent rate in England, Scotland and Wales reduced from 68.3%, 63.0% and 63.6% in April-June 2019 to 63.2%, 60.5% and 56.3% in April-June 2023.ConclusionsWhile the UK organ donation system shows signs of recovery from the COVID-19 pandemic, the number of eligible potential donors and consent rates remain below their pre-pandemic levels.

BackgroundKeeping the health workforce and the public informed about the latest evolving health information during a health emergency is critical to preventing, detecting and responding to infectious disease outbreaks or other health emergencies. Having a well-informed, ready, willing, and skilled workforce and an informed public can help save lives, reduce diseases and suffering, and minimize socio-economic loss in affected communities and countries. Providing "just in time" support and opportunities for learning in health emergencies is much needed for capacity building. In this paper, 'learning intervention' refers to the provision of ad-hoc, focused, or personalized training sessions with the goal of preparing the health workers for emergencies or filling specific knowledge or skill gaps. We refer to 'training methods' as instructional design strategies used to teach someone the necessary knowledge and skills to perform a task.MethodsWe conducted a scoping review to map and better understand what learning interventions and training methods have been used in different types of health emergencies and by whom. Studies were identified using six databases (Pubmed/Medline, Embase, Hinari, WorldCat, CABI and Web of Science) and by consulting with experts. Characteristics of studies were mapped and displayed and major topic areas were identified.ResultsOf the 319 records that were included, contexts most frequently covered were COVID-19, disasters in general, Ebola and wars. Four prominent topic areas were identified: 1) Knowledge acquisition, 2) Emergency plans, 3) Impact of the learning intervention, and 4) Training methods. Much of the evidence was based on observational methods with few trials, which likely reflects the unique context of each health emergency. Evolution of methods was apparent, particularly in virtual learning. Learning during health emergencies appeared to improve knowledge, general management of the situation, quality of life of both trainers and affected population, satisfaction and clinical outcomes.ConclusionThis is the first scoping review to map the evidence, which serves as a first step in developing urgently needed global guidance to further improve the quality and reach of learning interventions and training methods in this context.

Abstract Introduction Patient and public involvement (PPI) is increasingly seen as something that is integral to research and of importance to research funders. There is general recognition that PPI is the right thing to do for both moral and practical reasons. The aim of this review of reviews is to examine how PPI can be done ‘properly’ by looking at the evidence that exists from published reviews and assessing it against the UK Standards for Public Involvement in Research, as well as examining the specific features of population health research that can make PPI more challenging. Methods A review of reviews and development of best practice guidance was carried out following the 5-stage Framework Synthesis method. Results In total 31 reviews were included. There is a lack of current research or clarity around Governance and Impact when findings are mapped against UK Standards for Public Involvement in Research. It was also clear that there is little knowledge around PPI with under-represented groups. There are gaps in knowledge about how to ensure key specific attributes of population health research are addressed for PPI team members – particularly around how to deal with complexity and the data-driven nature of the research. Four tools were produced for researchers and PPI members to further improve their PPI activity within population health research and health research more generally, including a framework of recommended actions to address PPI in population health research, and guidance on integrating PPI based on the UK Standards for Public Involvement in Research. Conclusions Facilitating PPI in population health research is challenging due to the nature of this type of research and there is far less evidence on how to do PPI well in this context. The tools can help researchers identify key aspects of PPI that can be integrated when designing PPI within projects. Findings also highlight specific areas where more research or discussion is needed.

Objectives To synthesise qualitative research exploring patients’ perspectives, experiences and factors influencing their decision-making preferences when choosing or declining kidney transplantation.Design A qualitative evidence synthesis.Data sources Electronic databases were searched from 2000 to June 2021: PubMed, MEDLINE, CINAHL, Embase, PsycINFO, Web of Science, ProQuest Core Databases for Dissertations and Theses, and Google Scholar.Eligibility criteria Qualitative studies exploring and reporting decision-making preferences of people with kidney disease, which reported influencing factors when choosing or declining kidney transplantation, published in English from high-income and middle-income countries.Data extraction and synthesis Titles were screened against the inclusion criteria. Thematic synthesis was done with the use of the Critical Appraisal Skills Programme qualitative checklist to assess study quality, and assessment of confidence in the qualitative findings was done using the Grading of Recommendation, Assessment, Development and Evaluation Confidence in the Evidence from Reviews of Qualitative Research.Findings 37 studies from 11 countries reported the perspectives of 1366 patients with kidney disease. Six descriptive themes were developed: decisional preferences influenced patients’ readiness to pursue kidney transplantation, gathering sufficient information to support decision-making, navigating the kidney transplant assessment pathway, desire for kidney transplantation, opposed to kidney transplantation and uncertainties while waiting for the kidney transplant. A new enhanced theoretical model was developed to aid understanding of the complexities of decision-making in people with kidney disease, by integrating the Theory of Planned Behaviour and the Adaptive Decision Maker Framework to incorporate the novel findings.Conclusion The synthesis provides a better understanding of the extremely complex decision-making processes of people with kidney disease, which are aligned to their kidney transplantation preferences. Further research is needed to better understand the reasons for declining kidney transplantation, and to underpin development of personalised information, interventions and support for patients to make informed decisions when presented with kidney replacement options.PROSPERO registration number CRD42021272588.

Introduction There is growing recognition that in order to remain sustainable, the UK’s National Health Service must deliver the best patient outcomes within available resources. This focus on outcomes relative to cost is the basis of value-based healthcare (VBHC) and has led to interest in the recording of patient-reported outcome measures (PROMs) to measure patient perspectives on the impact of a health condition on their lives. Every health board in Wales is now required to collect PROMS as part of routine care. We will evaluate the VBHC programme implemented in a lead health board. The study aim is to understand what works about PROMs collection, for whom, in what contexts and why in a VBHC context. In addition, we will assess the social value of integrating PROMs collection into routine care.Methods and analysis A three-stage mixed-methods study comprising a realist evaluation integrated with social return on investment (SROI) analysis across four conditions; Parkinson’s disease, epilepsy, heart failure and cataract surgery. Workstream 1: Development of logic models, informed by a scoping review, documentary analysis, patient and public involvement (PPI), staff and key stakeholder engagement. Workstream 2: Realist evaluation building on multiple data sources from stages 1 to 3 to test and refine the programme theories that arise from the logic model development. Workstream 3: SROI analysis using interview data with patients, staff and carers, stakeholder and PPI engagement, anonymised routinely collected data, and questionnaires to populate a model that will explore the social value generated by the implementation of PROMs. Findings across stages will be validated with key stakeholders.Ethics and dissemination The study is approved by Wales Research Ethics Committee #5 (22/WA/0044). Outcomes will be shared with key stakeholders, published in peer-reviewed journals and presented at national and international conferences.This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) licence, which permits others to distribute, remix, adapt, build on this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial.

Abstract Context Too many people living with chronic kidney disease are opting for and starting on hospital‐based dialysis compared to a home‐based kidney replacement therapy. Dialysis services are becoming financially unsustainable. Objective This study aimed to assess the efficacy of coproductive research in chronic kidney disease service improvement to achieve greater sustainability. Design A 2‐year coproductive service improvement study was conducted with multiple stakeholders with the specific intention of maximizing engagement with the national health kidney services, patients and public. Setting and Participants A national health kidney service (3 health boards, 18 dialysis units), patients and families (n = 50), multidisciplinary teams including doctors, nurses, psychologists, social workers, and so forth (n = 68), kidney charities, independent dialysis service providers and wider social services were part of this study. Findings Coproductive research identified underutilized resources (e.g., patients on home dialysis and social services) and their potential, highlighted unmet social care needs for patients and families and informed service redesign. Education packages were reimagined to support the home dialysis agenda including opportunities for wider service input. The impacts of one size fits all approaches to dialysis on specialist workforce skills were made clearer and also professional, patient and public perceptions of key sustainability policies. Discussion and Conclusions Patient and key stakeholders mapped out new ways to link services to create more sustainable models of kidney health and social care. Maintaining principles of knowledge coproduction could help achieve financial sustainability and move towards more prudent adult chronic kidney disease services. Patient or Public Contribution Involved in developing research questions, study design, management and conduct, interpretation of evidence and dissemination.

Background : In the UK 1500 kidney transplants fail each year. 2% of living-donor transplants and 5% of deceased-donor transplants fail within a year of transplantation. Many decisions need to be made when a kidney transplant fails, including whether the transplant should be removed, and which immunosuppression medication should be stopped. There is limited evidence on which to base these decisions. We do not understand how decisions are made in the absence of good evidence, and there is national variation in practice. Aims : The overall aim is to develop a theory of patient and clinician transplant failure behaviours and decisions that will inform the design of a Randomised Controlled Trial (RCT) to evaluate treatments to optimise the management of transplant failure and improve outcomes for patients and their families. Methods : In a UK setting, we will develop a Constructivist Grounded theory using in-depth interviews with people over 18 years who are or have experienced kidney transplant failure, their families/close friends, and renal healthcare professional. Iterative purposive sampling of patients from different hospitals will be undertaken to achieve diversity with respect to age, sex, ethnicity, socioeconomic position, transplant type, and cause of transplant failure. Subsequent sampling will be theoretical, to test and develop hypotheses and theories being constructed. The sample size will be determined by reaching theoretical theme saturation with an anticipated minimum of 25–30 patients, 25–30 family members/close friends and 10–15 renal healthcare professionals. The research team will take a reflexive approach to make any influences or potential biases transparent. This knowledge will be used to develop the programme theory and design an RCT to evaluate treatments delivered at the right time in a patient’s journey, to improve experiences and outcomes for people with failing kidney transplants.

BackgroundValue-Based Healthcare (VBHC) focuses on the value of patient outcomes and is achieved by ensuring resources already available are managed to realise the best possible individual and population health outcomes. Patient reported outcome measures (PROMs) measure the impact of illnesses from the patient perspective. We conducted a scoping review to understand how PROMs were implemented and used, and their impact in the context of VBHC.MethodsArksey and O'Malley's overarching framework supplemented by principles from mixed-methods Framework Synthesis were used. CINAHL, Cochrane Library, EMBASE, MEDLINE, PsycINFO, Web of Science, Google Scholar and reference lists were searched. An a priori data extraction framework was created using the review question and objectives as key domains against which to extract data. Mixed-methods data were organised, integrated and preserved in original format and reported for each domain.ResultsForty-three studies were included with 60,200 participants. Few studies reported a well-developed programme theory and we found little robust evidence of effect. PROMs were universally considered to have the potential to increase patient satisfaction with treatment and services, enhance patient awareness of symptoms and self-management, and improve health outcomes such as quality of life and global health status. Evidence is currently limited on how PROMs work and how best to optimally implement PROMs to achieve the target outcome. Implementation challenges commonly prevented the realisation of optimal outcomes and patients generally needed better and clearer communication about why PROMs were being given and how they could optimally be used to support their own self-management.ConclusionPROMSs have yet to demonstrate their full potential in a VBHC context. Optimal PROMs implementation is poorly understood by clinicians and patients. Future studies should explore different models of PROM implementation and use within VBHC programmes to understand what works best and why for each specific context, condition, and population.

BackgroundLiving kidney organ donors offer a cost-effective alternative to deceased organ donation. They enable patients with life-threatening conditions to receive grafts that would otherwise not be available, thereby creating space for other patients waiting for organs and contributing to reducing overall waiting times for organs. There is an emerging consensus that an increase in living donation could contribute even more than deceased donation to reducing inequalities in organ donation between different population sub-groups in England. Increasing living donation is thus a priority for National Health Service Blood and Transplant (NHSBT) in the United Kingdom.MethodsUsing the random forest model, a machine learning (ML) approach, this study analyzed eight waves of repeated cross-sectional survey data collected from 2017 to 2021 (n = 14,278) as part of the organ donation attitudinal tracker survey commissioned by NHSBT in England to identify and help predict key factors that inform public intentions to become living donors.ResultsOverall, around 58.8% of the population would consider donating their kidney to a family member (50.5%), a friend (28%) or an unknown person (13.2%). The ML algorithm identified important factors that influence intentions to become a living kidney donor. They include, in reducing order of importance, support for organ donation, awareness of organ donation publicity campaigns, gender, age, occupation, religion, number of children in the household, and ethnic origin. Support for organ donation, awareness of public campaigns, and being younger were all positively associated with predicted propensity for living donation. The variable importance scores show that ethnic origin and religion were less important than the other variables in predicting living donor intention.ConclusionFactors influencing intentions to become a living donor are complex and highly individual in nature. Machine learning methods that allow for complex interactions between characteristics can be helpful in explaining these decisions. This work has identified important factors and subgroups that have higher propensity for living donation. Interventions should target both potential live donors and recipients. Research is needed to explore the extent to which these preferences are malleable to better understand what works and in which contexts to increase live organ donation.

BackgroundPreceded by a national media campaign, in May 2020, England switched to a soft opt-out system of organ donation which rests on the assumption that individuals meeting specific criteria have consented to organ donation unless they have expressed otherwise. We aimed to learn more about how the changes were communicated, how people responded and any discrepancies between key messages and how they were interpreted by the public.MethodsSummative content analysis of 286 stories and related reader-generated comments in leading UK online news sources (April 2019 to May 2021). Further detailed thematic analysis of 21 articles with reader-generated content, complemented by thematic content analysis coding of all 286 stories.ResultsMost media coverage on both organ donation and the law change was positive, with little variation over time or between publications. The importance of organ donation, benefits of the law change, and emotive stories (often involving children) of those who had donated an organ described as “superheroes” or those who had received organs as benefiting from a “miracle” were frequently cited. In contrast, reader-generated comments were markedly more negative, for example, focusing on loss of individual freedom and lack of trust in the organ donation system. Commentators wished to be able to choose who their organs were donated to, were dismissive and blaming towards minority ethnic groups, including undermining legitimate worries about the compatibility of organ donation with religious beliefs and end of life cultural norms, understanding and acceptance of brain-stem death and systemic racism. Misinformation including use of inflammatory language was common.ConclusionThe portrayal of donors and recipients as extraordinary is unlikely to help to normalise organ donation. Undermining legitimate concerns, in particular those from ethnic minorities, can alienate and encourage harmful misinformation in underrepresented groups. The discrepancies between the tone of the articles and the readers comments suggests a lack of trust across the public, health, policy and media outlets. Easily accessible, ongoing and tailored sources are needed to mitigate misinformation and disinformation and ensure key messages are better understood and accepted in order to realise the ambitions of soft opt-out organ donation policies.

Objective To determine intervention effects and synthesise qualitative research that explored women with or at high risk of kidney disease experiences of shared decision-making in relation to their reproductive health, family planning options and pregnancy.Design A systematic review of interventions and a qualitative evidence synthesis.Data sources We searched Cochrane, CINAHL, MEDLINE, Scopus, ProQuest, Elsevier, PubMed, ScienceDirect and Web of Science.Eligibility criteria Shared decision-making interventions and qualitative studies related to reproductive health involving women with or at high risk of kidney disease published from 1980 until January 2021 in English (clinical settings, global perspective).Data extraction and synthesis Titles were screened against the inclusion criteria and full-text articles were reviewed by the whole team. Framework synthesis was undertaken.Results We screened 1898 studies. No evidence-based interventions were identified. 18 qualitative studies were included, 11 kidney disease-specific studies and 7 where kidney disease was a common comorbidity. Women frequently felt unprepared and uninformed about their reproductive options. Conversations with healthcare professionals were commonly described as frustrating and unhelpful, often due to a perceived loss of autonomy and a mismatch in preferences and life goals. Examples of shared decision-making were rare. Kidney disease exacerbated societal expectations of traditional gender roles (eg, wife, mother, carer) including capability to have children and associated factors, for example, parenting, (sexual) relationships, body image and independent living (including financial barriers to starting a family). Local interventions were limited to types of counselling. A new health system model was developed to support new interventions.Conclusion There is a clear need to establish new interventions, test those already in development and develop new clinical guidance for the management of women with or at high risk of kidney disease in relation to their reproductive health, including options to preserve fertility earlier. Other health conditions with established personalised reproductive care packages, for example, cancer, could be used to benchmark kidney practice alongside the new model developed here.

Background: Although child mortality has decreased over the last few decades, around 4,500 infants and children die in the UK every year, many of whom require palliative care. There is, however, little evidence on paediatric end-of-life care services. The current National Institute for Health and Care Excellence (NICE) guidance provides recommendations about what should be offered, but these are based on low quality evidence. The ENHANCE study aims to identify and investigate the different models of existing end-of-life care provision for infants, children, and young people in the UK, including an assessment of the outcomes and experiences for children and parents, and the cost implications to families and healthcare providers. Methods: This mixed methods study will use three linked workstreams and a cross-cutting health economics theme to examine end-of-life care models in three exemplar clinical settings: infant, children and young adult cancer services (PTCs), paediatric intensive care units (PICUs), and neonatal units (NNUs). Workstream 1 (WS1) will survey current practice in each setting and will result in an outline of the different models of care used. WS2 is a qualitative comparison of the experiences of staff, parents and patients across the different models identified. WS3 is a quantitative assessment of the outcomes, resource use and costs across the different models identified. Discussion: Results from this study will contribute to an understanding of how end-of-life care can provide the greatest benefit for children at the end of their lives. It will also allow us to understand the likely benefits of additional funding in end-of-life care in terms of patient outcomes.

Objectives To explore how people with chronic kidney disease who are pre-dialysis, family members and healthcare professionals together navigate common shared decision-making processes and to assess how this impacts future treatment choice.Design Coproductive qualitative study, underpinned by the Making Good Decisions in Collaboration shared decision-model. Semistructured interviews with a purposive sample from February 2019 - January 2020. Interview data were analysed using framework analysis. Coproduction of logic models/roadmaps and recommendations.Setting Five Welsh kidney services.Participants 95 participants (37 patients, 19 family members and 39 professionals); 44 people supported coproduction (18 patients, 8 family members and 18 professionals).Findings Shared decision-making was too generic and clinically focused and had little impact on people getting onto home dialysis. Preferences of where, when and how to implement shared decision-making varied widely. Apathy experienced by patients, caused by lack of symptoms, denial, social circumstances and health systems issues made future treatment discussions difficult. Families had unmet and unrecognised needs, which significantly influenced patient decisions. Protocols containing treatment hierarchies and standards were understood by professionals but not translated for patients and families. Variation in dialysis treatment was discussed to match individual lifestyles. Patients and professionals were, however, defaulting to the perceived simplest option. It was easy for patients to opt for hospital-based treatments by listing important but easily modifiable factors.Conclusions Shared decision-making processes need to be individually tailored with more attention on patients who could choose a home therapy but select a different option. There are critical points in the decision-making process where changes could benefit patients. Patients need to be better educated and their preconceived ideas and misconceptions gently challenged. Healthcare professionals need to update their knowledge in order to provide the best advice and guidance. There needs to be more awareness of the costs and benefits of the various treatment options when making decisions.

Abstract Background Co‐production of research into public health services has yet to demonstrate tangible benefits. Few studies have reported the impact of co‐production on research outcomes. The previous studies of organ donation have identified challenges in engaging with public organizations responsible, gaining ethical approval for sensitive studies with the recently bereaved and difficulty in recruiting bereaved family members who were approached about organ donation. Objective To address these challenges, we designed the first large co‐productive observational study to evaluate implementation of a new system of organ donation in Wales. This paper outlines the co‐productive strategies that were designed to overcome known methodological challenges and reports what impact they had on resolving these challenges. Design Two‐year co‐produced study with multiple stakeholders with the specific intention of maximizing engagement with the National Health Service arm in Wales responsible for organ donation, and recruitment of bereaved family members whose perspectives are essential but commonly absent from studies. Setting and participants NHS Blood and Transplant, Welsh Government and multiple patient and public representatives who served as co‐productive partners with the research team. Results Co‐productive strategies enabled a smooth passage through four different ethics processes within the 10‐week time frame, family member recruitment targets to be surpassed, sharing of routinely collected data on 100% of potential organ donor cases and development of further research capacity and capability in a critically under researched area. Discussion and conclusion Although expensive and time consuming, co‐production was effective and added value to research processes and study outcomes.

Abstract In an attempt to improve organ donation rates, some countries are considering moving from “opt‐in” systems where citizens must express their willingness to be an organ donor, to “opt‐out” systems where consent is presumed unless individuals have expressed their wishes otherwise, by, for example, joining an “opt‐out” register. In Wales—a part of the United Kingdom—the devolved government recently legislated for an “opt‐out” system. For the change to be effective, a public awareness campaign was critical to the policy's success. Using quantitative and qualitative content analysis, we explored media coverage of the change to better understand the relationship between the state, policy actors, media and the public when such policy changes take place. Our findings illustrate how a state communication campaign can effectively set the media agenda within which we saw a degree of interdependency created with the state using the media to promote policy, and the media relying on the state for credible information. Yet we also found that the media is not uncritical and observed how it uses its autonomy to influence policy setting. Over the period of study, we found that a change in tone and view towards deemed consent organ donation has taken place in the media. However, while this may influence or reflect public attitudes, it is yet to be seen whether the media campaign translates into behavioural change that will result in increases in organ donations.

Abstract Background Qualitative evidence synthesis is increasingly used alongside reviews of effectiveness to inform guidelines and other decisions. To support this use, the GRADE-CERQual approach was developed to assess and communicate the confidence we have in findings from reviews of qualitative research. One component of this approach requires an appraisal of the methodological limitations of studies contributing data to a review finding. Diverse critical appraisal tools for qualitative research are currently being used. However, it is unclear which tool is most appropriate for informing a GRADE-CERQual assessment of confidence. Methodology We searched for tools that were explicitly intended for critically appraising the methodological quality of qualitative research. We searched the reference lists of existing methodological reviews for critical appraisal tools, and also conducted a systematic search in June 2016 for tools published in health science and social science databases. Two reviewers screened identified titles and abstracts, and then screened the full text of potentially relevant articles. One reviewer extracted data from each article and a second reviewer checked the extraction. We used a best-fit framework synthesis approach to code checklist criteria from each identified tool and to organise these into themes. Results We identified 102 critical appraisal tools: 71 tools had previously been included in methodological reviews, and 31 tools were identified from our systematic search. Almost half of the tools were published after 2010. Few authors described how their tool was developed, or why a new tool was needed. After coding all criteria, we developed a framework that included 22 themes. None of the tools included all 22 themes. Some themes were included in up to 95 of the tools. Conclusion It is problematic that researchers continue to develop new tools without adequately examining the many tools that already exist. Furthermore, the plethora of tools, old and new, indicates a lack of consensus regarding the best tool to use, and an absence of empirical evidence about the most important criteria for assessing the methodological limitations of qualitative research, including in the context of use with GRADE-CERQual.

Abstract Background Wales introduced a soft opt-out organ donation system on 1st December 2015 with the aim of improving consent rates. In the first 18 months consent rates improved but the difference could not solely be attributed to the soft opt-out system when compared with similar improvements in consent rates in other UK nations. Methods We conducted an 18 month post-intervention qualitative process evaluation involving 88 family members of 60/211 potential organ donor cases, and 19 professionals. Views and experiences of Specialist Nurses in Organ Donation who implemented the new system and family members who were involved in decision making were collected to see how their respective behaviours impacted on implementation. Data collection included interviews, focus groups and qualitative questionnaire data. Results Implementation was considered a success by Specialist Nurses in Organ Donation. The bespoke retraining programme and responsive approach to addressing initial implementation issues were identified as examples of best practice. Specialist Nurses in Organ Donation were valued by family members. Six implementation issues had an impact on consent rates – the media campaign had gaps, the system was more complex, challenges in changing professional behaviours, inability to obtain the required standard of evidence from family members to overturn a donation decision, increased complexity of consent processes, and additional health systems issues. Conclusion This is the first comprehensive process evaluation of implementing a soft opt-out system of organ donation. Specific elements of good implementation practice (such as investment in the retraining programme and the responsiveness of Specialist Nurses in Organ Donation and managers to feedback) were identified. The key message is that despite retraining, nursing practice did not radically change overnight to accommodate the new soft opt-out system. Policy makers and health service managers should not assume that nurses simply need more time to implement the soft-out as intended. Additional responsive modification of processes, ongoing training and support is required to help with implementation as originally intended. Scotland, England and the Netherlands are introducing soft opt-out systems. There is an opportunity to learn from initial implementation in Wales, by acknowledging gaps, good practice and opportunities to further improve processes and nursing practices.

Abstract Background It is not known which attributes of care are valued the most by those who experience hospice services. Such knowledge is integral to service development as it facilitates opportunities for continuous improvement of hospice care provision. The objectives of this mixed-studies systematic review were to explore patients’ and their family carer views and experiences, to determine what they valued about adult hospice care in the UK. Methods ASSIA, PubMed, CINAHL and PsycINFO were searched from inception, up until March 2017 to identify qualitative, quantitative, and mixed-methods studies. Four additional searching techniques supplemented the main search and grey literature was included. A three-stage mixed-method systematic review was conducted with a sequential exploratory design. Thematic synthesis was used with qualitative data, followed by a narrative summary of the quantitative data. The qualitative and quantitative syntheses were then juxtaposed within a matrix to produce an overarching synthesis. Results Thirty-four studies highlighted that what patients and carers valued was generally context specific and stemmed from an amalgamation of hospice service components, which both individually and collectively contributed to improvements in quality of life. When the syntheses of qualitative and quantitative studies were viewed in isolation, the value placed on services remained relatively consistent, with some discrepancies evident in service availability. These were commonly associated with geographical variations, as well as differences in service models and timeframes. Through an overarching synthesis of the qualitative and quantitative evidence, however, notable variations and a more nuanced account of what people valued and why were more prominent, specifically in relation to a lack of social support for carers, disparate access to essential services, the underrepresentation of patients with a non-cancer diagnosis, and the dissatisfaction with the range of services provided. Conclusion Review findings strengthen the existing evidence base and illuminates the underpinning elements of hospice care most valued by patients and their families. With large disparities in the availability of services, however, the underrepresentation of patients with non-malignant diseases and the limited evidence base demonstrating the adequate addressment of the social needs of carers, there continues to be considerable gaps that warrants further research.

Abstract Background Decision making in health and social care requires robust syntheses of both quantitative and qualitative evidence. Meta-ethnography is a seven-phase methodology for synthesising qualitative studies. Developed in 1988 by sociologists in education Noblit and Hare, meta-ethnography has evolved since its inception; it is now widely used in healthcare research and is gaining popularity in education research. The aim of this article is to provide up-to-date, in-depth guidance on conducting the complex analytic synthesis phases 4 to 6 of meta-ethnography through analysis of the latest methodological evidence. Methods We report findings from a methodological systematic review conducted from 2015 to 2016. Fourteen databases and five other online resources were searched. Expansive searches were also conducted resulting in inclusion of 57 publications on meta-ethnography conduct and reporting from a range of academic disciplines published from 1988 to 2016. Results Current guidance on applying meta-ethnography originates from a small group of researchers using the methodology in a health context. We identified that researchers have operationalised the analysis and synthesis methods of meta-ethnography – determining how studies are related (phase 4), translating studies into one another (phase 5), synthesising translations (phase 6) and line of argument synthesis - to suit their own syntheses resulting in variation in methods and their application. Empirical research is required to compare the impact of different methods of translation and synthesis. Some methods are potentially better at preserving links with the context and meaning of primary studies, a key principle of meta-ethnography. A meta-ethnography can and should include reciprocal and refutational translation and line of argument synthesis, rather than only one of these, to maximise the impact of its outputs. Conclusion The current work is the first to articulate and differentiate the methodological variations and their application for different purposes and represents a significant advance in the understanding of the methodological application of meta-ethnography.

Abstract Aims The aim of this study was to provide guidance to improve the completeness and clarity of meta-ethnography reporting. Background Evidence-based policy and practice require robust evidence syntheses which can further understanding of people’s experiences and associated social processes. Meta-ethnography is a rigorous seven-phase qualitative evidence synthesis methodology, developed by Noblit and Hare. Meta-ethnography is used widely in health research, but reporting is often poor quality and this discourages trust in and use of its findings. Meta-ethnography reporting guidance is needed to improve reporting quality. Design The eMERGe study used a rigorous mixed-methods design and evidence-based methods to develop the novel reporting guidance and explanatory notes. Methods The study, conducted from 2015 to 2017, comprised of: (1) a methodological systematic review of guidance for meta-ethnography conduct and reporting; (2) a review and audit of published meta-ethnographies to identify good practice principles; (3) international, multidisciplinary consensus-building processes to agree guidance content; (4) innovative development of the guidance and explanatory notes. Findings Recommendations and good practice for all seven phases of meta-ethnography conduct and reporting were newly identified leading to 19 reporting criteria and accompanying detailed guidance. Conclusion The bespoke eMERGe Reporting Guidance, which incorporates new methodological developments and advances the methodology, can help researchers to report the important aspects of meta-ethnography. Use of the guidance should raise reporting quality. Better reporting could make assessments of confidence in the findings more robust and increase use of meta-ethnography outputs to improve practice, policy, and service user outcomes in health and other fields. This is the first tailored reporting guideline for meta-ethnography. This article is being simultaneously published in the following journals: Journal of Advanced Nursing, Psycho-oncology, Review of Education, and BMC Medical Research Methodology.

Qualitative evidence syntheses (QES) have increased in prominence and profile over the last decade as a discrete set of methodologies to undertake systematic reviews of primary qualitative research in health and social care and in education. The findings from a qualitative evidence synthesis can enable a richer interpretation of a particular phenomenon, set of circumstances, or experiences than single primary qualitative research studies can achieve. Qualitative evidence synthesis methods were developed in response to an increasing demand from health and social professionals, policy makers, guideline developers and educationalists for review evidence that goes beyond “what works” afforded by systematic reviews of effectiveness. The increasing interest in the synthesis of qualitative research has led to methodological developments documented across a plethora of texts and journal articles. This “State of the Method” paper aims to bring together these methodological developments in one place, contextualizing advances in methods with exemplars to support readers in making choices in approach to a synthesis and aid understanding. The paper clarifies what a “qualitative evidence synthesis” is and explores its role, purpose and development. It details the kind of questions a QES can explore, the processes associated with a QES, including the methods for synthesis. The rational and methods for integrating a QES with systematic reviews of effectiveness are also detailed. Finally approaches reporting and recognition of what a “good” or rigorous QES look like are provided.

Background: The number of young adults with complex health-care needs due to life-limiting conditions/complex physical disability has risen significantly over the last 15 years, as more children now survive into adulthood. The transition from children to adult services may disrupt provision of essential respite/short break care for this vulnerable population, but the impact on young adults, families and providers is unclear. Aim: To review the evidence on respite care provision for young adults (aged 18–40 years) with complex health-care needs, provide an evidence gap analysis and develop a conceptual framework for respite care. Design: A two-stage mixed-methods systematic review, including a knowledge map of respite care and an evidence review of policy, effectiveness, cost-effectiveness and experience. Data sources: Electronic databases and grey/unpublished literature were searched from 2002 to September 2019. The databases searched included Cumulative Index to Nursing and Allied Health Literature, MEDLINE, EMBASE, PsycINFO, Applied Social Sciences Index and Abstracts, Health Management Information Consortium, PROSPERO, Turning Research into Practice, COnNECT+, British Nursing Index, Web of Science, Social Care Online, the National Institute for Health Research Journals Library, Cochrane Effective Practice and Organisation of Care specialist register, databases on The Cochrane Library and international clinical trials registers. Additional sources were searched using the CLUSTER (Citations, Lead authors, Unpublished materials, Scholar search, Theories, Early examples, Related projects) approach and an international ‘call for evidence’. Methods and analysis: Multiple independent reviewers used the SPICE (Setting, Perspective, Intervention/phenomenon of interest, Comparison, Evaluation) framework to select and extract evidence for each stage, verified by a third reviewer. Study/source characteristics and outcomes were extracted. Study quality was assessed using relevant tools. Qualitative evidence was synthesised using a framework approach and UK policy was synthesised using documentary content analysis. GRADE-CERQual (Grading of Recommendations Assessment, Development and Evaluation-Confidence in the Evidence from Reviews of Qualitative Research) was used to assess confidence in the evidence. Logic models developed for each type of respite care constituted the conceptual framework. Results: We identified 69 sources (78 records) from 126,267 records. The knowledge map comprised the following types of respite care: residential, home based, day care, community, leisure/social provision, funded holidays and emergency. Seven policy intentions included early transition planning and prioritising respite care according to need. No evidence was found on effectiveness and cost-effectiveness. Qualitative evidence focused largely on residential respite care. Facilitators of accessible/acceptable services included trusted and valued relationships, independence and empowerment of young adults, peer social interaction, developmental/age-appropriate services and high standards of care. Barriers included transition to adult services, paperwork, referral/provision delay and travelling distance. Young adults from black, Asian and minority ethnic populations were under-represented. Poor transition, such as loss of or inappropriate services, was contrary to statutory expectations. Potential harms included stress and anxiety related to safe care, frustration and distress arising from unmet needs, parental exhaustion, and a lack of opportunities to socialise and develop independence. Limitations: No quantitative or mixed-methods evidence was found on effectiveness or cost-effectiveness of respite care. There was limited evidence on planned and emergency respite care except residential. Conclusions: Policy intentions are more comprehensively met for young people aged

Introduction The mental health and well-being of children and young people who have been in care (ie, care-experienced) are a priority. There are a range of interventions aimed at addressing these outcomes, but the international evidence-base remains ambiguous. There is a paucity of methodologically robust systematic reviews of intervention effectiveness, with few considering the contextual conditions under which evaluations were conducted. This is important in understanding the potential transferability of the evidence-base across contexts. The present systematic review will adopt a complex systems perspective to synthesise evidence reporting evaluations of mental health and well-being interventions for care-experienced children and young people. It will address impact, equity, cost-effectiveness, context, implementation and acceptability. Stakeholder consultation will prioritise a programme theory, and associated intervention, that may progress to further development and evaluation in the UK.Methods and analysis We will search 16 bibliographic databases from 1990 to June 2020. Supplementary searching will include citation tracking, author recommendation, and identification of evidence clusters relevant to included evaluations. The eligible population is children and young people (aged ≤25 years) with experience of being in care. Outcomes are (1) mental, behavioural or neurodevelopmental disorders; (2) subjective well-being; (3) self-harm; suicidal ideation; suicide. Study quality will be appraised with methodologically appropriate tools. We will construct a taxonomy of programme theories and intervention types. Thematic synthesis will be used for qualitative data reporting context, implementation and acceptability. If appropriate, meta-analysis will be conducted with outcome and economic data. Convergent synthesis will be used to integrate syntheses of qualitative and quantitative data.Ethics and dissemination We have a comprehensive strategy for engagement with care-experienced children and young people, carers and social care professionals. Dissemination will include academic and non-academic publications and conference presentations. Ethical approval from Cardiff University’s School of Social Sciences REC will be obtained if necessary.PROSPERO registration number CRD42020177478.

Background: One-fifth of all disabled children have mobility limitations. Early provision of powered mobility for very young children (aged

Objectives To identify the shared research priorities of patients, caregivers and multidisciplinary renal health and social care professionals across Wales for integrated renal health and social care in Wales.Design Research priority setting exercise adapted from the James Lind Alliance national priority setting partnership framework in UK healthcare.Setting Two workshops: one in North Wales with patients, caregivers and multidisciplinary renal health and social care professionals and one in South Wales with the Welsh Renal Clinical Network (commissioners of renal services in Wales). Additional input provided from stakeholders via email correspondence and face to face communications.Participants Academics n=14, patients n=16, family/carers n=6, multidisciplinary renal healthcare professionals n=40, local authority councils n=3, renal charities n=6 wider third sector organisations n=8, renal industries n=4, Welsh government social care n=3, renal service commissioners n=8.Results 38 research priority questions grouped into 10 themes were agreed. The themes included: (1) integrating health and social care, (2) education, (3) acute kidney injury, (4) chronic kidney disease and cardiovascular disease, (5) transplantation, (6) dialysis, (7) personalised medicines, (8) cross-cutting priorities, (9) specific social contexts and (10) transitional services and children. Research questions were broad and covered a range of health and social care topics. Patient and professional perspectives broadly overlapped. Research priority setting activities revealed gaps in knowledge in overall service provision and potential areas for service improvement.Conclusions Mapping priorities in health services and social care highlighted the research needed to support renal health services delivery and commissioning in Wales.

Abstract Background The GRADE-CERQual (Confidence in Evidence from Reviews of Qualitative research) approach has been developed by the GRADE (Grading of Recommendations Assessment, Development and Evaluation) Working Group. The approach has been developed to support the use of findings from qualitative evidence syntheses in decision-making, including guideline development and policy formulation. CERQual includes four components for assessing how much confidence to place in findings from reviews of qualitative research (also referred to as qualitative evidence syntheses): (1) methodological limitations, (2) coherence, (3) adequacy of data and (4) relevance. This paper is part of a series providing guidance on how to apply CERQual and focuses on CERQual’s relevance component. Methods We developed the relevance component by searching the literature for definitions, gathering feedback from relevant research communities and developing consensus through project group meetings. We tested the CERQual relevance component within several qualitative evidence syntheses before agreeing on the current definition and principles for application. Results When applying CERQual, we define relevance as the extent to which the body of data from the primary studies supporting a review finding is applicable to the context (perspective or population, phenomenon of interest, setting) specified in the review question. In this paper, we describe the relevance component and its rationale and offer guidance on how to assess relevance in the context of a review finding. This guidance outlines the information required to assess relevance, the steps that need to be taken to assess relevance and examples of relevance assessments. Conclusions This paper provides guidance for review authors and others on undertaking an assessment of relevance in the context of the CERQual approach. Assessing the relevance component requires consideration of potentially important contextual factors at an early stage in the review process. We expect the CERQual approach, and its individual components, to develop further as our experiences with the practical implementation of the approach increase.

Abstract Background The GRADE-CERQual (Confidence in Evidence from Reviews of Qualitative research) approach has been developed by the GRADE (Grading of Recommendations Assessment, Development and Evaluation) Working Group. The approach has been developed to support the use of findings from qualitative evidence syntheses in decision-making, including guideline development and policy formulation. CERQual includes four components for assessing how much confidence to place in findings from reviews of qualitative research (also referred to as qualitative evidence syntheses): (1) methodological limitations, (2) coherence, (3) adequacy of data and (4) relevance. This paper is part of a series providing guidance on how to apply CERQual and focuses on a probable fifth component, dissemination bias. Given its exploratory nature, we are not yet able to provide guidance on applying this potential component of the CERQual approach. Instead, we focus on how dissemination bias might be conceptualised in the context of qualitative research and the potential impact dissemination bias might have on an overall assessment of confidence in a review finding. We also set out a proposed research agenda in this area. Methods We developed this paper by gathering feedback from relevant research communities, searching MEDLINE and Web of Science to identify and characterise the existing literature discussing or assessing dissemination bias in qualitative research and its wider implications, developing consensus through project group meetings, and conducting an online survey of the extent, awareness and perceptions of dissemination bias in qualitative research. Results We have defined dissemination bias in qualitative research as a systematic distortion of the phenomenon of interest due to selective dissemination of studies or individual study findings. Dissemination bias is important for qualitative evidence syntheses as the selective dissemination of qualitative studies and/or study findings may distort our understanding of the phenomena that these syntheses aim to explore and thereby undermine our confidence in these findings. Dissemination bias has been extensively examined in the context of randomised controlled trials and systematic reviews of such studies. The effects of potential dissemination bias are formally considered, as publication bias, within the GRADE approach. However, the issue has received almost no attention in the context of qualitative research. Because of very limited understanding of dissemination bias and its potential impact on review findings in the context of qualitative evidence syntheses, this component is currently not included in the GRADE-CERQual approach. Conclusions Further research is needed to establish the extent and impacts of dissemination bias in qualitative research and the extent to which dissemination bias needs to be taken into account when we assess how much confidence we have in findings from qualitative evidence syntheses.

Abstract Background The GRADE-CERQual (Grading of Recommendations Assessment, Development and Evaluation-Confidence in Evidence from Reviews of Qualitative research) approach has been developed by the GRADE working group. The approach has been developed to support the use of findings from qualitative evidence syntheses in decision-making, including guideline development and policy formulation. CERQual includes four components for assessing how much confidence to place in findings from reviews of qualitative research (also referred to as qualitative evidence syntheses): (1) methodological limitations, (2) relevance, (3) coherence and (4) adequacy of data. This paper is part of a series providing guidance on how to apply CERQual and focuses on CERQual’s coherence component. Methods We developed the coherence component by searching the literature for definitions, gathering feedback from relevant research communities and developing consensus through project group meetings. We tested the CERQual coherence component within several qualitative evidence syntheses before agreeing on the current definition and principles for application. Results When applying CERQual, we define coherence as how clear and cogent the fit is between the data from the primary studies and a review finding that synthesises that data. In this paper, we describe the coherence component and its rationale and offer guidance on how to assess coherence in the context of a review finding as part of the CERQual approach. This guidance outlines the information required to assess coherence, the steps that need to be taken to assess coherence and examples of coherence assessments. Conclusions This paper provides guidance for review authors and others on undertaking an assessment of coherence in the context of the CERQual approach. We suggest that threats to coherence may arise when the data supporting a review finding are contradictory, ambiguous or incomplete or where competing theories exist that could be used to synthesise the data. We expect the CERQual approach, and its individual components, to develop further as our experiences with the practical implementation of the approach increase.

Abstract Background The GRADE-CERQual (Confidence in Evidence from Reviews of Qualitative research) approach has been developed by the GRADE (Grading of Recommendations Assessment, Development and Evaluation) Working Group. The approach has been developed to support the use of findings from qualitative evidence syntheses in decision making, including guideline development and policy formulation. CERQual includes four components for assessing how much confidence to place in findings from reviews of qualitative research (also referred to as qualitative evidence syntheses): (1) methodological limitations, (2) coherence, (3) adequacy of data and (4) relevance. This paper is part of a series providing guidance on how to apply CERQual and focuses on making an overall assessment of confidence in a review finding and creating a CERQual Evidence Profile and a CERQual Summary of Qualitative Findings table. Methods We developed this guidance by examining the methods used by other GRADE approaches, gathering feedback from relevant research communities and developing consensus through project group meetings. We then piloted the guidance on several qualitative evidence syntheses before agreeing on the approach. Results Confidence in the evidence is an assessment of the extent to which a review finding is a reasonable representation of the phenomenon of interest. Creating a summary of each review finding and deciding whether or not CERQual should be used are important steps prior to assessing confidence. Confidence should be assessed for each review finding individually, based on the judgements made for each of the four CERQual components. Four levels are used to describe the overall assessment of confidence: high, moderate, low or very low. The overall CERQual assessment for each review finding should be explained in a CERQual Evidence Profile and Summary of Qualitative Findings table. Conclusions Structuring and summarising review findings, assessing confidence in those findings using CERQual and creating a CERQual Evidence Profile and Summary of Qualitative Findings table should be essential components of undertaking qualitative evidence syntheses. This paper describes the end point of a CERQual assessment and should be read in conjunction with the other papers in the series that provide information on assessing individual CERQual components.

Abstract Background The GRADE-CERQual (Confidence in Evidence from Reviews of Qualitative research) approach has been developed by the GRADE (Grading of Recommendations Assessment, Development and Evaluation) Working Group. The approach has been developed to support the use of findings from qualitative evidence syntheses in decision-making, including guideline development and policy formulation. CERQual includes four components for assessing how much confidence to place in findings from reviews of qualitative research (also referred to as qualitative evidence syntheses): (1) methodological limitations, (2) coherence, (3) adequacy of data and (4) relevance. This paper is part of a series providing guidance on how to apply CERQual and focuses on CERQual’s methodological limitations component. Methods We developed the methodological limitations component by searching the literature for definitions, gathering feedback from relevant research communities and developing consensus through project group meetings. We tested the CERQual methodological limitations component within several qualitative evidence syntheses before agreeing on the current definition and principles for application. Results When applying CERQual, we define methodological limitations as the extent to which there are concerns about the design or conduct of the primary studies that contributed evidence to an individual review finding. In this paper, we describe the methodological limitations component and its rationale and offer guidance on how to assess methodological limitations of a review finding as part of the CERQual approach. This guidance outlines the information required to assess methodological limitations component, the steps that need to be taken to assess methodological limitations of data contributing to a review finding and examples of methodological limitation assessments. Conclusions This paper provides guidance for review authors and others on undertaking an assessment of methodological limitations in the context of the CERQual approach. More work is needed to determine which criteria critical appraisal tools should include when assessing methodological limitations. We currently recommend that whichever tool is used, review authors provide a transparent description of their assessments of methodological limitations in a review finding. We expect the CERQual approach and its individual components to develop further as our experiences with the practical implementation of the approach increase.

Abstract Background The GRADE-CERQual (Confidence in Evidence from Reviews of Qualitative research) approach has been developed by the GRADE (Grading of Recommendations Assessment, Development and Evaluation) working group. The approach has been developed to support the use of findings from qualitative evidence syntheses in decision-making, including guideline development and policy formulation. CERQual includes four components for assessing how much confidence to place in findings from reviews of qualitative research (also referred to as qualitative evidence syntheses): (1) methodological limitations; (2) coherence; (3) adequacy of data; and (4) relevance. This paper is part of a series providing guidance on how to apply CERQual and focuses on CERQual’s adequacy of data component. Methods We developed the adequacy of data component by searching the literature for definitions, gathering feedback from relevant research communities and developing consensus through project group meetings. We tested the CERQual adequacy of data component within several qualitative evidence syntheses before agreeing on the current definition and principles for application. Results When applying CERQual, we define adequacy of data as an overall determination of the degree of richness and the quantity of data supporting a review finding. In this paper, we describe the adequacy component and its rationale and offer guidance on how to assess data adequacy in the context of a review finding as part of the CERQual approach. This guidance outlines the information required to assess data adequacy, the steps that need to be taken to assess data adequacy, and examples of adequacy assessments. Conclusions This paper provides guidance for review authors and others on undertaking an assessment of adequacy in the context of the CERQual approach. We approach assessments of data adequacy in terms of the richness and quantity of the data supporting each review finding, but do not offer fixed rules regarding what constitutes sufficiently rich data or an adequate quantity of data. Instead, we recommend that this assessment is made in relation to the nature of the finding. We expect the CERQual approach, and its individual components, to develop further as our experiences with the practical implementation of the approach increase.

Abstract The GRADE-CERQual (‘Confidence in the Evidence from Reviews of Qualitative research’) approach provides guidance for assessing how much confidence to place in findings from systematic reviews of qualitative research (or qualitative evidence syntheses). The approach has been developed to support the use of findings from qualitative evidence syntheses in decision-making, including guideline development and policy formulation. Confidence in the evidence from qualitative evidence syntheses is an assessment of the extent to which a review finding is a reasonable representation of the phenomenon of interest. CERQual provides a systematic and transparent framework for assessing confidence in individual review findings, based on consideration of four components: (1) methodological limitations, (2) coherence, (3) adequacy of data, and (4) relevance. A fifth component, dissemination (or publication) bias, may also be important and is being explored. As with the GRADE (Grading of Recommendations Assessment, Development, and Evaluation) approach for effectiveness evidence, CERQual suggests summarising evidence in succinct, transparent, and informative Summary of Qualitative Findings tables. These tables are designed to communicate the review findings and the CERQual assessment of confidence in each finding. This article is the first of a seven-part series providing guidance on how to apply the CERQual approach. In this paper, we describe the rationale and conceptual basis for CERQual, the aims of the approach, how the approach was developed, and its main components. We also outline the purpose and structure of this series and discuss the growing role for qualitative evidence in decision-making. Papers 3, 4, 5, 6, and 7 in this series discuss each CERQual component, including the rationale for including the component in the approach, how the component is conceptualised, and how it should be assessed. Paper 2 discusses how to make an overall assessment of confidence in a review finding and how to create a Summary of Qualitative Findings table. The series is intended primarily for those undertaking qualitative evidence syntheses or using their findings in decision-making processes but is also relevant to guideline development agencies, primary qualitative researchers, and implementation scientists and practitioners.

Abstract Background Unintended repeat conceptions can result in emotional, psychological and educational harm to young women, often with enduring implications for their life chances. This study aimed to identify which young women are at the greatest risk of repeat unintended pregnancies; which interventions are effective and cost-effective; and what are the barriers to and facilitators for the uptake of these interventions. Methods We conducted a mixed-methods systematic review which included meta-analysis, framework synthesis and application of realist principles, with stakeholder input and service user feedback to address this. We searched 20 electronic databases, including MEDLINE, Excerpta Medica database, Applied Social Sciences Index and Abstracts and Research Papers in Economics, to cover a broad range of health, social science, health economics and grey literature sources. Searches were conducted between May 2013 and June 2014 and updated in August 2015. Results Twelve randomised controlled trials (RCTs), two quasi-RCTs, 10 qualitative studies and 53 other quantitative studies were identified. The RCTs evaluated psychosocial interventions and an emergency contraception programme. The primary outcome was repeat conception rate: the event rate was 132 of 308 (43%) in the intervention group versus 140 of 289 (48%) for the control group, with a non-significant risk ratio (RR) of 0.92 [95% confidence interval (CI) 0.78–1.08]. Four studies reported subsequent birth rates: 29 of 237 (12%) events for the intervention arm versus 46 out of 224 (21%) for the control arm, with an RR of 0.60 (95% CI 0.39–0.93). Many repeat conceptions occurred in the context of poverty, low expectations and aspirations and negligible opportunities. Qualitative and realist evidence highlighted the importance of context, motivation, future planning and giving young women a central and active role in the development of new interventions. Conclusions Little or no evidence for the effectiveness or cost-effectiveness of any of the interventions to reduce repeat pregnancy in young women was found. Qualitative and realist evidence helped to explain gaps in intervention design that should be addressed. More theory-based, rigorously evaluated programmes need to be developed to reduce unintended repeat pregnancy in young women. Trial registration PROSPERO, CRD42012003168 . Cochrane registration number: i = fertility/0068

Abstract Objective The objective of this research project was to evaluate the validity of proxy health-related quality of life measures in the context of paediatric mobility impairment. Accurate health-related quality of life data is essential for quality-adjusted life year calculation; a key outcome in economic evaluation. Thirteen child-parent dyads (13 children with mobility impairments, 13 parent proxies) were asked to complete a range of outcome measures (EQ-5D-Y, VAS and HUI2/3) relating to the child’s health. The relationship between respondent outcomes was examined using tests of respondent type effect (Wilcoxon signed-rank), correlation (Spearman’s rank-order) and agreement (Bland–Altman plots). Results Parent proxies significantly undervalued the health-related quality of life of their mobility-impaired children: children rated their health-related quality of life higher than their parents by proxy on all measures. The VAS had the highest overall mean score for children and proxies (79.50 [SD = 15.01] and 75.77 [SD = 14.70] respectively). Child and proxy results were significantly different (p

Abstract Background Health interventions fall along a spectrum from simple to more complex. There is wide interest in methods for reviewing ‘complex interventions’, but few transparent approaches for assessing intervention complexity in systematic reviews. Such assessments may assist review authors in, for example, systematically describing interventions and developing logic models. This paper describes the development and application of the intervention Complexity Assessment Tool for Systematic Reviews (iCAT_SR), a new tool to assess and categorise levels of intervention complexity in systematic reviews. Methods We developed the iCAT_SR by adapting and extending an existing complexity assessment tool for randomized trials. We undertook this adaptation using a consensus approach in which possible complexity dimensions were circulated for feedback to a panel of methodologists with expertise in complex interventions and systematic reviews. Based on these inputs, we developed a draft version of the tool. We then invited a second round of feedback from the panel and a wider group of systematic reviewers. This informed further refinement of the tool. Results The tool comprises ten dimensions: (1) the number of active components in the intervention; (2) the number of behaviours of recipients to which the intervention is directed; (3) the range and number of organizational levels targeted by the intervention; (4) the degree of tailoring intended or flexibility permitted across sites or individuals in applying or implementing the intervention; (5) the level of skill required by those delivering the intervention; (6) the level of skill required by those receiving the intervention; (7) the degree of interaction between intervention components; (8) the degree to which the effects of the intervention are context dependent; (9) the degree to which the effects of the interventions are changed by recipient or provider factors; (10) and the nature of the causal pathway between intervention and outcome. Dimensions 1–6 are considered ‘core’ dimensions. Dimensions 7–10 are optional and may not be useful for all interventions. Conclusions The iCAT_SR tool facilitates more in-depth, systematic assessment of the complexity of interventions in systematic reviews and can assist in undertaking reviews and interpreting review findings. Further testing of the tool is now needed.

IntroductionThe number of young adults with complex healthcare needs due to life-limiting conditions/complex physical disability has risen significantly as children with complex conditions survive into adulthood. Respite care and short breaks are an essential service, however, needs often go unmet after the transition to adult services, leading to a significant impact on the life expectancy and quality of life for this population. We aim to identify, appraise and synthesise relevant evidence to explore respite care and short breaks provision for this population, and to develop a conceptual framework for understanding service models.Methods and analysisA mixed-methods systematic review conducted in two stages: (1) knowledge map and (2) evidence review. We will comprehensively search multiple electronic databases; use the Citations, Lead authors, Unpublished materials, Google Scholar, Theories, Early examples, and Related projects (CLUSTER) approach, search relevant websites and circulate a ‘call for evidence’. Using the setting, perspective, intervention/phenomenon of interest, comparison and evaluation framework, two reviewers will independently select evidence for inclusion into a knowledge map and subsequent evidence review, extract data relating to study and population characteristics, methods and outcomes; and assess the quality of evidence. A third reviewer will arbitrate where necessary.Evidence will be synthesised using the following approaches: quantitative (narratively/conducting meta-analyses where appropriate); qualitative (framework approach); policy and guidelines (documentary analysis informed approach). An overall, integrated synthesis will be created using a modified framework approach. We will use Grading of Recommendations Assessment, Development and Evaluation (GRADE)/GRADE-Confidence in the Evidence from Reviews of Qualitative Research to assess the strength and confidence of the synthesised evidence. Throughout, we will develop a conceptual framework to articulate how service models work in relation to context and setting.Ethics and disseminationEthical approval is not required as this is a systematic review. We will present our work in academic journals, at appropriate conferences; we will disseminate findings across networks using a range of media. Steering and advisory groups were established to ensure findings are shared widely and in accessible formats.PROSPERO registration numberCRD42018088780.

Background: Meta-ethnography is a commonly used methodology for qualitative evidence synthesis. Research has identified that the quality of reporting of published meta-ethnographies is often poor and this has limited the utility of meta-ethnography findings to influence policy and practice. Objective: To develop guidance to improve the completeness and clarity of meta-ethnography reporting. Methods/design: The meta-ethnography reporting guidance (eMERGe) study followed the recommended approach for developing health research reporting guidelines and used a systematic mixed-methods approach. It comprised (1) a methodological systematic review of guidance in the conduct and reporting of meta-ethnography; (2) a review and audit of published meta-ethnographies, along with interviews with meta-ethnography end-users, to identify good practice principles; (3) a consensus workshop and two eDelphi (Version 1, Duncan E, Swinger K, University of Stirling, Stirling, UK) studies to agree guidance content; and (4) the development of the guidance table and explanatory notes. Results: Results from the methodological systematic review and the audit of published meta-ethnographies revealed that more guidance was required around the reporting of all phases of meta-ethnography conduct and, in particular, the synthesis phases 4–6 (relating studies, translating studies into one another and synthesising translations). Following the guidance development process, the eMERGe reporting guidance was produced, comprising 19 items grouped into the seven phases of meta-ethnography. Limitations: The finalised guidance has not yet been evaluated in practice; therefore, it is not possible at this stage to comment on its utility. However, we look forward to evaluating its uptake and usability in the future. Conclusions: The eMERGe reporting guidance has been developed following a rigorous process in line with guideline development recommendations. The guidance is intended to improve the clarity and completeness of reporting of meta-ethnographies, and to facilitate use of the findings within the guidance to inform the design and delivery of services and interventions in health, social care and other fields. The eMERGe project developed a range of training materials to support use of the guidance, which is freely available at www.emergeproject.org (accessed 26 March 2018). Meta-ethnography is an evolving qualitative evidence synthesis methodology and future research should refine the guidance to accommodate future methodological developments. We will also investigate the impact of the eMERGe reporting guidance with a view to updating the guidance. Study registration: This study is registered as PROSPERO CRD42015024709 for the stage 1 systematic review. Funding: The National Institute for Health Research Health Services and Delivery Research programme.

Young people age 14-25 years with chronic kidney disease have been identified as generally having poor health outcomes and are a high-risk group for kidney transplant loss due in part to poor self-management. This raises a key question as to what happens during transition from child to adult services? This paper presents a mixed-method systematic review of health and social care evidence concerning young people with chronic kidney disease transitioning from child to adult health and social care services. Quantitative and qualitative evidence were synthesised in streams followed by an overarching synthesis. Literature searches (2000 to March 2017) were conducted using Pubmed, BioMed Central and Cochrane Library, grey literature sources ZETOC, .gov.uk, third sector organisations, NHS Evidence, SCIE, TRIP, Opengrey. Snowball searching was conducted in the databases Ovid, CINAHL, ISI Web of Science, Scopus and Google Scholar. Of 3,125 records screened, 60 texts were included. We found that while strategies to support transition contained consistent messages, they supported the principle of a health-dominated pathway. Well-being is mainly defined and measured in clinical terms and the transition process is often presented as a linear, one-dimensional conduit. Individual characteristics, along with social, familial and societal relationships are rarely considered. Evidence from young people and their families highlights transition as a zone of conflict between independence and dependency with young people feeling powerless on one hand and overwhelmed on the other. We found few novel interventions and fewer that had been evaluated. Studies were rarely conducted by allied health and social care professionals (e.g. renal social workers and psychologists) as part of multi-disciplinary renal teams. We conclude that there is a lack of good evidence to inform providers of health and social care services about how best to meet the needs of this small but vulnerable cohort.

Wheelchairs for children with impaired mobility provide health, developmental and psychosocial benefits, however there is limited understanding of how mobility aids affect the health-related quality of life of children with impaired mobility. Preference-based health-related quality of life outcome measures are used to calculate quality-adjusted life years; an important concept in health economics. The aim of this research was to understand how young wheelchair users and their parents define health-related quality of life in relation to mobility impairment and wheelchair use.The sampling frame was children with impaired mobility (≤18 years) who use a wheelchair and their parents. Data were collected through semi-structured face-to-face interviews conducted in participants' homes. Qualitative framework analysis was used to analyse the interview transcripts. An a priori thematic coding framework was developed. Emerging codes were grouped into categories, and refined into analytical themes. The data were used to build an understanding of how children with impaired mobility define health-related quality of life in relation to mobility impairment, and to assess the applicability of two standard measures of health-related quality of life.Eleven children with impaired mobility and 24 parents were interviewed across 27 interviews. Participants defined mobility-related quality of life through three distinct but interrelated concepts: 1) participation and positive experiences; 2) self-worth and feeling fulfilled; 3) health and functioning. A good degree of consensus was found between child and parent responses, although there was some evidence to suggest a shift in perception of mobility-related quality of life with child age.Young wheelchair users define health-related quality of life in a distinct way as a result of their mobility impairment and adaptation use. Generic, preference-based measures of health-related quality of life lack sensitivity in this population. Development of a mobility-related quality of life outcome measure for children is recommended.

Patellofemoral pain syndrome (PFPS) is a very common knee condition with various aetiologies. Because of the nebulous factors of the syndrome, physiotherapists often find it difficult to assess and treat these patients. The aim of this study was to identify the clinical assessment tool that can differentiate PFPS patients from patients with other lower limb conditions. Fifty-two patients from the National Health System (26 with PFPS and 26 with other lower limb conditions) took part in this study. They underwent a series of strength, flexibility, and stress tests. Their pain levels were also recorded. The results showed that among the various clinical tests, only the hip flexion component of the Thomas test was able to differentiate between the two groups. In addition, the stress test showed that the PFPS group could not recover their gluteal muscle strength in the same way the group with the other lower limb conditions did. The Lower Extremity Functional Scale was found to be more able to differentiate between the two groups than the Anterior Knee Pain Scale. This study has shown that it is difficult to find specific clinical tests to diagnose PFPS. More research is needed in this important area.