Understanding how children and young people with chronic non-cancer pain and their families experience living with pain, pain management and services: a meta-ethnography.

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.

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.

The implementation, use and impact of patient reported outcome measures in value-based healthcare programmes: A scoping review.

BACKGROUND: Value-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. METHODS: Arksey 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. RESULTS: Forty-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. CONCLUSION: PROMSs 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.

A meta-ethnography of how children and young people with chronic non-cancer pain and their families experience and understand their condition, pain services, and treatments.

BACKGROUND: Chronic non-cancer pain in childhood is widespread, affecting 20% to 35% of children and young people worldwide. For a sizeable number of children, chronic non-cancer pain has considerable negative impacts on their lives and quality of life, and leads to increased use of healthcare services and medication. In many countries, there are few services for managing children's chronic non-cancer pain, with many services being inadequate. Fourteen Cochrane Reviews assessing the effects of pharmacological, psychological, psychosocial, dietary or physical activity interventions for managing children's chronic non-cancer pain identified a lack of high-quality evidence to inform pain management. To design and deliver services and interventions that meet the needs of patients and their families, we need to understand how children with chronic non-cancer pain and their families experience pain, their views of services and treatments for chronic pain, and which outcomes are important to them. OBJECTIVES: 1. To synthesise qualitative studies that examine the experiences and perceptions of children with chronic non-cancer pain and their families regarding chronic non-cancer pain, treatments and services to inform the design and delivery of health and social care services, interventions and future research. 2. To explore whether our review findings help to explain the results of Cochrane Reviews of intervention effects of treatments for children's chronic non-cancer pain. 3. To determine if programme theories and outcomes of interventions match children and their families' views of desired treatments and outcomes. 4. To use our findings to inform the selection and design of patient-reported outcome measures for use in chronic non-cancer pain studies and interventions and care provision to children and their families. The review questions are: 1. How do children with chronic non-cancer pain and their families conceptualise chronic pain? 2. How do children with chronic non-cancer pain and their families live with chronic pain? 3. What do children with chronic non-cancer pain and their families think of how health and social care services respond to and manage their child's chronic pain? 4. What do children with chronic non-cancer pain and their families conceptualise as 'good' chronic pain management and what do they want to achieve from chronic pain management interventions and services? SEARCH METHODS: Review strategy: we comprehensively searched 12 bibliographic databases including MEDLINE, CINAHL, PsycInfo and grey literature sources, and conducted supplementary searches in 2020. We updated the database searches in September 2022. SELECTION CRITERIA: To identify published and unpublished qualitative research with children aged 3 months to 18 years with chronic non-cancer pain and their families focusing on their perceptions, experiences and views of chronic pain, services and treatments. The final inclusion criteria were agreed with a patient and public involvement group of children and young people with chronic non-cancer pain and their families. DATA COLLECTION AND ANALYSIS: We conducted a qualitative evidence synthesis using meta-ethnography, a seven-phase, systematic, interpretive, inductive methodology that takes into account the contexts and meanings of the original studies. We assessed the richness of eligible studies and purposively sampled rich studies ensuring they addressed the review questions. Cochrane Qualitative Methods Implementation Group guidance guided sampling. We assessed the methodological limitations of studies using the Critical Appraisal Skills Programme tool. We extracted data on study aims, focus, characteristics and conceptual findings from study reports using NVivo software. We compared these study data to determine how the studies related to one another and grouped studies by pain conditions for synthesis. 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. Sampling, organising studies for synthesis, and analysis and interpretation involved our patient and public involvement group who contributed throughout the conduct of the review. We used the GRADE-CERQual (Confidence in the Evidence from Reviews of Qualitative research) approach to assess our confidence in each review finding. We used a matrix approach to integrate our findings with existing Cochrane Reviews on treatment effectiveness for children's chronic non-cancer pain. MAIN RESULTS: We synthesised 43 studies sampled from 170 eligible studies reported in 182 publications. Included studies involved 633 participants. GRADE-CERQual assessments of findings were mostly high (n = 21, 58%) or moderate (n = 12, 33%) confidence with three (8%) low or very low confidence. Poorly managed, moderate or severe chronic non-cancer pain had profound adverse impacts on family dynamics and relationships; family members' emotions, well-being, autonomy and sense of self-identity; parenting strategies; friendships and socialising; children's education and future employment prospects; and parental employment. Most children and parents understood chronic non-cancer pain as having an underlying biological cause and wanted curative treatment. However, families had difficulties seeking and obtaining support from health services to manage their child's pain and its impacts. Children and parents felt that healthcare professionals did not always listen to their experiences and expertise, or believe the child's pain. Some families repeatedly visited health services seeking a diagnosis and cure. Over time, some children and families gave up hope of effective treatment. Outcomes measured within trials and Cochrane Reviews of intervention effects did not include some outcomes of importance to children and families, including impacts of pain on the whole family and absence of pain. Cochrane Reviews have mainly neglected a holistic biopsychosocial approach, which specifies the interrelatedness of biological, psychological and social aspects of illness, when selecting outcome measures and considering how chronic pain management interventions work. AUTHORS' CONCLUSIONS: We had high or moderate confidence in the evidence contributing to most review findings. Further research, especially into families' experiences of treatments and services, could strengthen the evidence for low or very low confidence findings. Future research should also explore families' experiences in low- to middle-income contexts; of pain treatments including opioid use in children, which remains controversial; and of social care services. We need development and testing of family-centred interventions and services acceptable to families. Future trials of children's chronic non-cancer pain interventions should include family-centred outcomes.