Health outcomes in Deaf signing populations: A systematic review.

OBJECTIVES: (i) To identify peer reviewed publications reporting the mental and/or physical health outcomes of Deaf adults who are sign language users and to synthesise evidence; (ii) If data available, to analyse how the health of the adult Deaf population compares to that of the general population; (iii) to evaluate the quality of evidence in the identified publications; (iv) to identify limitations of the current evidence base and suggest directions for future research. DESIGN: Systematic review. DATA SOURCES: Medline, Embase, PsychINFO, and Web of Science. ELIGIBILITY CRITERIA FOR SELECTING STUDIES: The inclusion criteria were Deaf adult populations who used a signed language, all study types, including methods-focused papers which also contain results in relation to health outcomes of Deaf signing populations. Full-text articles, published in peer-review journals were searched up to 13th June 2023, published in English or a signed language such as ASL (American Sign Language). DATA EXTRACTION: Supported by the Rayyan systematic review software, two authors independently reviewed identified publications at each screening stage (primary and secondary). A third reviewer was consulted to settle any disagreements. Comprehensive data extraction included research design, study sample, methodology, findings, and a quality assessment. RESULTS: Of the 35 included studies, the majority (25 out of 35) concerned mental health outcomes. The findings from this review highlighted the inequalities in health and mental health outcomes for Deaf signing populations in comparison with the general population, gaps in the range of conditions studied in relation to Deaf people, and the poor quality of available data. CONCLUSIONS: Population sample definition and consistency of standards of reporting of health outcomes for Deaf people who use sign language should be improved. Further research on health outcomes not previously reported is needed to gain better understanding of Deaf people's state of health.

Making Use of Technology to Improve Stated Preference Studies.

The interest in quantifying stated preferences for health and healthcare continues to grow, as does the technology available to support and improve health preference studies. Technological advancements in the last two decades have implications and opportunities for preference researchers designing, administering, analysing, interpreting and applying the results of stated preference surveys. In this paper, we summarise selected technologies and how these can benefit a preference study. We discuss empirical evaluations of the technology in preference research, with examples from health where possible. The technologies reviewed include serious games, virtual reality, eye tracking, innovative formats and decision aids with values clarification components. We conclude with a critical reflection on the benefits and limitations of implementing (often costly) technology alongside stated preference studies.

A service mapping exercise of four health and social care staff mental health and wellbeing services, Resilience Hubs, to describe health service provision and interventions.

BACKGROUND: NHS England funded 40 Mental Health and Wellbeing Hubs to support health and social care staff affected by the COVID-19 pandemic. We aimed to document variations in how national guidance was adapted to the local contexts of four Hubs in the North of England. METHODS: We used a modified version of Price's (2019) service mapping methodology. Service level data were used to inform the analysis. A mapping template was adapted from a range of tools, including the European Service Mapping Schedule, and reviewed by Hub leads. Key data included service model; staffing; and interventions. Data were collected between March 2021 - March 2022 by site research assistants. Findings were accuracy-checked by Hub leads, and a logic model developed to theorise how the Hubs may effect change. RESULTS: Hub goals and service models closely reflected guidance; offering: proactive outreach; team-based support; clinical assessment; onward referral, and rapid access to mental health support (in-house and external). Implementation reflected a service context of a client group with high mental health need, and high waiting times at external mental health services. Hubs were predominantly staffed by experienced clinicians, to manage these mental health presentations and organisational working. Formulation-based psychological assessment and the provision of direct therapy were not core functions of the NHS England model, however all Hubs incorporated these adaptations into their service models in response to local contexts, such as extensive waiting lists within external services, and/or client presentations falling between gaps in existing service provision. Finally, a standalone clinical records system was seen as important to reassure Hub users of confidentiality. Other more nuanced variation depended on localised contexts. CONCLUSION: This study provides a map for setting up services, emphasising early understandings of how new services will integrate within existing systems. Local and regional contexts led to variation in service configuration. Whilst additional Hub functions are supported by available literature, further research is needed to determine whether these functions should comprise essential components of staff wellbeing services moving forward. Future research should also determine the comparative effectiveness of service components, and the limits of permissible variation. STUDY REGISTRATION: researchregistry6303.

Advances in Addressing Patient Heterogeneity in Economic Evaluation: A Review of the Methods Literature.

Cost-effectiveness analyses commonly use population or sample averages, which can mask key differences across subgroups and may lead to suboptimal resource allocation. Despite there being several new methods developed over the last decade, there is no recent summary of what methods are available to researchers. This review sought to identify advances in methods for addressing patient heterogeneity in economic evaluations and to provide an overview of these methods. A literature search was conducted using the Econlit, Embase and MEDLINE databases to identify studies published after 2011 (date of a previous review on this topic). Eligible studies needed to have an explicit methodological focus, related to how patient heterogeneity can be accounted for within a full economic evaluation. Sixteen studies were included in the review. Methodologies were varied and included regression techniques, model design and value of information analysis. Recent publications have applied methodologies more commonly used in other fields, such as machine learning and causal forests. Commonly noted challenges associated with considering patient heterogeneity included data availability (e.g., sample size), statistical issues (e.g., risk of false positives) and practical factors (e.g., computation time). A range of methods are available to address patient heterogeneity in economic evaluation, with relevant methods differing according to research question, scope of the economic evaluation and data availability. Researchers need to be aware of the challenges associated with addressing patient heterogeneity (e.g., data availability) to ensure findings are meaningful and robust. Future research is needed to assess whether and how methods are being applied in practice.

Motiv8: a study protocol for a cluster-randomised feasibility trial of a weight management intervention for adults with severe mental illness in secure forensic services.

INTRODUCTION: People with severe mental illness have physical comorbidities which result in significant reductions in quality of life and premature mortality. Effective interventions are required that are suitable for people in secure forensic mental health services. We conducted pilot work of a multidisciplinary weight management intervention (Motiv8) which showed improvements in physical and mental health and high levels of satisfaction. We aim to test the feasibility of Motiv8 under cluster randomised conditions, with an aim to investigate the acceptability, feasibility and potential effectiveness of this intervention to supplement standard secure care. METHODS AND ANALYSIS: A randomised waitlist-controlled feasibility trial of a lifestyle intervention (Motiv8) + TAU compared with TAU (+ Motiv8 waitlist) for adults on secure mental health units will be conducted. Thirty-two people (4 cohorts) will be recruited from secure services in Greater Manchester Mental Health NHS Foundation Trust. Participants will be randomly allocated to Motiv8 or TAU + Motiv8 waitlist. All participants will receive Motiv8 during the trial. Assessor-blinded physical/mental health and lifestyle assessments will be conducted at baseline, 10 weeks (post-intervention/waitlist), and after 12 weeks (post-waitlist intervention/follow-up). Motiv8 is a multidisciplinary intervention including exercise sessions, cooking/nutrition classes, physical health education, psychology sessions, sleep hygiene, peer support and medication review by pharmacy. A nested qualitative study will be conducted with a subsample of participants (n = 10) to explore their experiences of taking part. The analysis will focus on feasibility outcomes and tabulated success indicators of the study (e.g. Recruitment rates, retention rates, follow-up retention and response rates, attendance at sessions, the experience of involvement in the trial and delivery of the intervention, assessment of safety, development of a manualised intervention). Thematic analysis will be conducted through qualitative interviews. The analysis will aim to inform the development of a definitive trial. ETHICS AND DISSEMINATION: The trial has been granted ethical approval from the NHS Health Research Authority and adopted onto the UK Clinical Research Network Portfolio. Findings will be disseminated via peer-reviewed publications, professional and public networks, conferences and clinical services. TRIAL REGISTRATION: ISRCTN13539285.

Generating EQ-5D-3L health utility scores from the Edinburgh Postnatal Depression Scale: a perinatal mapping study.

BACKGROUND: Perinatal depression (PND) describes depression experienced by parents during pregnancy or in the first year after a baby is born. The EQ-5D instrument (a generic measure of health status) is not often collected in perinatal research, however disease-specific measures, such as the Edinburgh Postnatal Depression Scale (EPDS) are widely used. Mapping can be used to estimate generic health utility index values from disease-specific measures like the EPDS. OBJECTIVE: To develop a mapping algorithm to estimate EQ-5D utility index values from the EPDS. METHODS: Patient-level data from the BaBY PaNDA study (English observational cohort study) provided 1068 observations with paired EPDS and EQ-5D (3-level version; EQ-5D-3L) responses. We compared the performance of six alternative regression model types, each with four specifications of covariates (EPDS score and age: base, squared, and cubed). Model performance (ability to predict utility values) was assessed by ranking mean error, mean absolute error, and root mean square error. Algorithm performance in 3 external datasets was also evaluated. RESULTS: There was moderate correlation between EPDS score and utility values (coefficient: - 0.42). The best performing model type was a two-part model, followed by ordinary least squared. Inclusion of squared and cubed covariates improved model performance. Based on graphs of observed and predicted utility values, the algorithm performed better when utility was above 0.6. CONCLUSIONS: This direct mapping algorithm allows the estimation of health utility values from EPDS scores. The algorithm has good external validity but is likely to perform better in samples with higher health status.

An economic evaluation of universal and targeted case-finding strategies for identifying antenatal depression: a model-based analysis comparing common case-finding instruments.

Half of women with depression in the perinatal period are not identified in routine care, and missed cases reflect inequalities in other areas of maternity care. Case finding (screening) for depression in pregnant women may be a cost-effective strategy to improve identification, and targeted case finding directs finite resources towards the greatest need. We compared the cost-effectiveness of three case-finding strategies: no case finding, universal (all pregnant women), and targeted (only pregnant women with risk factors for antenatal depression, i.e. history of anxiety/depression, age < 20 years, and adverse life events). A decision tree model was developed to represent case finding (at around 20 weeks gestation) and subsequent treatment for antenatal depression (up to 40 weeks gestation). Costs include case finding and treatment. Health benefits are measured as quality-adjusted life years (QALYs). The sensitivity and specificity of case-finding instruments and prevalence and severity of antenatal depression were estimated from a cohort study of pregnant women. Other model parameters were derived from published literature and expert consultation. The most cost-effective case-finding strategy was a two-stage strategy comprising the Whooley questions followed by the PHQ-9. The mean costs were £52 (universal), £61 (no case finding), and £62 (targeted case finding). Both case-finding strategies improve health compared with no case finding. Universal case finding is cost-saving. Costs associated with targeted case finding are similar to no case finding, with greater health gains, although targeted case finding is not cost-effective compared with universal case finding. Universal case finding for antenatal depression is cost-saving compared to no case finding and more cost-effective than targeted case finding.

Protocol of a feasibility randomised controlled trial of Empowered Conversations: training family carers to enhance their relationships and communication with people living with dementia.

Background: Communication difficulties can cause frustration, low mood, and stress for people living with dementia and their carer. Carers should be offered training on adapting their communication skills. However, it is not common for skills-based education to examine emotional aspects of care and the effect of dementia on relationships. The Empowered Conversations (EC) training course was developed in response to a gap in service provision and has been adapted to a virtual format (Zoom). It addresses the specific psychological, relationship, and communication needs of informal and family dementia carers. The primary aim of the study is to investigate the feasibility of conducting a multi-centre randomised controlled evaluation trial of EC. Secondary aims include exploring the acceptability of delivering the intervention online and examining the optimum way of establishing cost-effectiveness. Methods: The feasibility trial uses a pragmatic data-collector blind parallel two-group RCT design with two arms (EC intervention plus treatment as usual, and treatment as usual waitlist control). There will be a 2:1 allocation in favour of the EC-training intervention arm. 75 participants will complete baseline outcome measures exploring their role as a carer, including their physical and mental health, attitudes to caring, quality of life, and use of health and social care services. These will be repeated after six-months. Participants allocated to the treatment group who complete the course will be invited to participate in a qualitative interview discussing their experience of EC. Conclusions: The study will investigate recruitment pathways (including facilitators and barriers to recruitment), estimate retention levels and response rates to questionnaires, obtain additional evidence regarding proof of concept, and consider the most appropriate primary outcome measures and methods for evaluating cost-effectiveness. The results of the feasibility study will be used to inform the development of a multicentre randomised controlled trial in the United Kingdom. Registration: ISRCTN15261686 (02/03/2022).

There are 700,000 family and informal carers for people living with dementia in the UK alone. Sixty-four percent of informal carers in England say they have limited support for the range of psychological and social needs they experience. It can be difficult to keep communicating well due to thinking and memory changes that caused by dementia. This can lead to frustration, low-mood and stress for both people living with dementia and their carers. The 6-session online Empowered Conversations course is designed to enable carers to maintain and improve good communication and relationships with those they support. Course facilitators are trained to provide specific communication techniques, ways of managing conflicts, and working with difficult emotions. The course has been tried out over the last 4-years and changes made. Feedback from informal carers indicates it is in an optimum form and we are ready to test it further in a large trial. Before this is done, it is necessary to complete a smaller 'feasibility' trial to check whether such a larger trial is possible. This article explains how the feasibility trial will be carried out. Our 'feasibility' trial will check several things. We want to make sure that carers would be willing to have an only 66% chance of receiving the course straight away, because it is essential to have a comparison group. The remaining 33% of carers would be offered the course 6-months later. We want to ensure that our design is good enough to identify any improvement in carers' well-being, relationships and communication. We will also ask carers to take part in a one-to-one interview about their experiences of the course, including their views on the course being delivered on Zoom.

A randomised controlled trial of clinical and cost-effectiveness of the PASS Plus intervention for young children with autism spectrum disorder in New Delhi, India: study protocol for the COMPASS trial.

BACKGROUND: Autism Spectrum Disorder (ASD) is a neurodevelopmental disability affecting at least 5 million children in South Asia. Majority of these children are without access to evidence-based care. The UK Pre-school Autism Communication Therapy (PACT) is the only intervention to have shown sustained impact on autism symptoms. It was systematically adapted for non-specialist community delivery in South Asia, as the 'Parent-mediated Autism Social Communication Intervention for non-Specialists (PASS)' and extended 'PASS Plus' interventions. RCTs of both showed feasibility, acceptability and positive effect on parent and child dyadic outcomes. METHODS: The Communication-centred Parent-mediated treatment for Autism Spectrum Disorder in South Asia (COMPASS) trial is now a scale-up two-centre, two-arm single (rater) blinded random allocation parallel group study of the PASS Plus intervention in addition to treatment as usual (TAU) compared to TAU alone, plus health economic evaluation embedded in the India health system. Two hundred forty children (approximately 120 intervention/120 TAU) with ASD aged 2-9 years will be recruited from two tertiary care government hospitals in New Delhi, India. Accredited Social Health Activists will be one of the intervention delivery agents. Families will undertake up to 12 communication sessions over 8 months and will be offered the Plus modules which address coexisting problems. The trial's primary endpoint is at 9 months from randomisation, with follow-up at 15 months. The primary outcome is autism symptom severity; secondary outcomes include parent-child communication, child adaptation, quality of life and parental wellbeing. Primary analysis will follow intention-to-treat principles using linear mixed model regressions with group allocation and repeated measures as random effects. The cost-effectiveness analysis will use a societal perspective over the 15-month period of intervention and follow-up. DISCUSSION: If clinically and cost-effective, this programme will fill an important gap of scalable interventions delivered by non-specialist health workers within the current care pathways for autistic children and their families in low-resource contexts. The programme has been implemented through the COVID-19 pandemic when restrictions were in place; intervention delivery and evaluation processes have been adapted to address these conditions. TRIAL REGISTRATION: ISRCTN; ISRCTN21454676 ; Registered 22 June 2018.

An economic evaluation of targeted case-finding strategies for identifying postnatal depression: A model-based analysis comparing common case-finding instruments.

BACKGROUND: Half of women with postnatal depression (PND) are not identified in routine care. We aimed to estimate the cost-effectiveness of PND case-finding in women with risk factors for PND. METHODS: A decision tree was developed to represent the one-year costs and health outcomes associated with case-finding and treatment for PND. The sensitivity and specificity of case-finding instruments, and prevalence and severity of PND, for women with ≥1 PND risk factor were estimated from a cohort of postnatal women. Risk factors were history of anxiety/depression, age < 20 years, and adverse life events. Other model parameters were derived from published literature and expert consultation. Case-finding for high-risk women only was compared with no case-finding and universal case-finding. RESULTS: More than half of the cohort had one or more PND risk factor (57.8 %; 95 % CI 52.7 %-62.7 %). The most cost-effective case-finding strategy was the Edinburgh Postnatal Depression Scale with a cut-off of ≥10 (EPDS-10). Among high-risk women, there is a high probability that EPDS-10 case-finding for PND is cost-effective compared to no case-finding (78.5 % at a threshold of £20,000/QALY), with an ICER of £8146/QALY gained. Universal case-finding is even more cost-effective at £2945/QALY gained (versus no case-finding). There is a greater health improvement with universal rather than targeted case-finding. LIMITATIONS: The model includes costs and health benefits for mothers in the first year postpartum, the broader (e.g. families, societal) and long-term impacts are also important. CONCLUSIONS: Universal PND case-finding is more cost-effective than targeted case-finding which itself is more cost-effective than not case-finding.

A Systematic Review of Methods to Incorporate External Evidence into Trial-Based Survival Extrapolations for Health Technology Assessment.

BACKGROUND: External evidence is commonly used to inform survival modeling for health technology assessment (HTA). While there are a range of methodological approaches that have been proposed, it is unclear which methods could be used and how they compare. PURPOSE: This review aims to identify, describe, and categorize established methods to incorporate external evidence into survival extrapolation for HTA. DATA SOURCES: Embase, MEDLINE, EconLit, and Web of Science databases were searched to identify published methodological studies, supplemented by hand searching and citation tracking. STUDY SELECTION: Eligible studies were required to present a novel extrapolation approach incorporating external evidence (i.e., data or information) within survival model estimation. DATA EXTRACTION: Studies were classified according to how the external evidence was integrated as a part of model fitting. Information was extracted concerning the model-fitting process, key requirements, assumptions, software, application contexts, and presentation of comparisons with, or validation against, other methods. DATA SYNTHESIS: Across 18 methods identified from 22 studies, themes included use of informative prior(s) (n = 5), piecewise (n = 7), and general population adjustment (n = 9), plus a variety of "other" (n = 8) approaches. Most methods were applied in cancer populations (n = 13). No studies compared or validated their method against another method that also incorporated external evidence. LIMITATIONS: As only studies with a specific methodological objective were included, methods proposed as part of another study type (e.g., an economic evaluation) were excluded from this review. CONCLUSIONS: Several methods were identified in this review, with common themes based on typical data sources and analytical approaches. Of note, no evidence was found comparing the identified methods to one another, and so an assessment of different methods would be a useful area for further research.HighlightsThis review aims to identify methods that have been used to incorporate external evidence into survival extrapolations, focusing on those that may be used to inform health technology assessment.We found a range of different approaches, including piecewise methods, Bayesian methods using informative priors, and general population adjustment methods, as well as a variety of "other" approaches.No studies attempted to compare the performance of alternative methods for incorporating external evidence with respect to the accuracy of survival predictions. Further research investigating this would be valuable.

Responsiveness to change in health status of the EQ-5D in patients treated for depression and anxiety.

BACKGROUND: The EQ-5D is a commonly used generic measure of health but evidence on its responsiveness to change in mental health is limited. This study aimed to explore the responsiveness of the five-level version of the instrument, the EQ-5D-5 L, in patients receiving treatment for depression and anxiety. METHODS: Patient data (N = 416) were collected at baseline and at end of treatment in an observational study in a Norwegian outpatient clinic. Patients were adults of working age (18-69 years) and received protocol-based metacognitive or cognitive therapy for depression or anxiety according to diagnosis. Responsiveness in the EQ-5D was compared to change in the Beck Depression Inventory-II (BDI-II) and the Beck Anxiety Inventory (BAI). Effect sizes (Cohen's d), Standardised response mean (SRM), and Pearson's correlation were calculated. Patients were classified as "Recovered", "Improved", or "Unchanged" during treatment using the BDI-II and the BAI. ROC analyses determined whether the EQ-5D could correctly classify patient outcomes. RESULTS: Effect sizes were large for the BAI, the BDI-II, the EQ-5D value and the EQ VAS, ranging from d = 1.07 to d = 1.84. SRM were also large (0.93-1.67). Pearson's correlation showed strong agreement between change scores of the EQ-5D value and the BDI-II (rs -0.54) and moderate between the EQ-5D value and the BAI (rs -0.43). The EQ-5D consistently identified "Recovered" patients versus "Improved" or "Unchanged" in the ROC analyses with AUROC ranging from 0.72 to 0.84. CONCLUSION: The EQ-5D showed good agreement with self-reported symptom change in depression and anxiety, and correctly identified recovered patients. These findings indicate that the EQ-5D may be appropriately responsive to change in patients with depression and anxiety disorders, although replication in other clinical samples is needed.

The EQ-5D is a questionnaire that people fill in to report their subjective health. It is often used in clinics or hospitals to better understand how patients are affected by their illnesses, and if their health improves after treatment. For this information to be trustworthy, we need to verify how accurately the EQ-5D measures health for the particular patients we want to use it with. This is often done by comparing EQ-5D scores with scores from other questionnaires. For example, if we want to use the EQ-5D with a group of patients with depression, we compare the scores of the EQ-5D with scores from questionnaires that are commonly used to measure depression symptoms.In this study, we compared the scores of the EQ-5D with scores from questionnaires measuring symptoms of depression and anxiety. Their performances were similar, and the EQ-5D scores could also correctly identify which patients had recovered during treatment. This implies that the EQ-5D can be a useful tool for understanding the impact of depression and anxiety and can help in decision-making regarding these patients.

An Investigation into the Relationship Between Choice of Model Structure and How to Adjust for Subsequent Therapies Using a Case Study in Oncology.

BACKGROUND: A common challenge in health technology assessments (HTAs) of cancer treatments is how subsequent therapy use within the trial follow-up may influence cost-effectiveness model outcomes. Although overall survival (OS) is often a key driver of model results, there are no guidelines to advise how to adjust for this potential confounding, with different approaches available dependent on the model structure. OBJECTIVE: We compared a partitioned survival analysis (PartSA) with a semi-Markov multi-state model (MSM) structure, with and without attempts to adjust for the impact of subsequent therapies on OS using a case study describing outcomes for people with relapsed/refractory multiple myeloma. METHODS: Both model structures included three health states: pre-progression, progressed disease and death. Three traditional crossover methods were considered within the context of the PartSA, whereas for the MSM, the probability of post-progression death was pooled across arms. Impacts on the model incremental cost-effectiveness ratio (ICER) were recorded. RESULTS: The unadjusted PartSA produced an ICER of £623,563, and after adjustment yielded an ICER range of £381,340-£386,907. The unadjusted MSM produced an ICER of £1,283,780. Adjusting OS in the MSM resulted in an ICER of £345,486. CONCLUSIONS: The simplicity of the PartSA is lost when the decision problem becomes more complex (for example, when OS data are confounded by subsequent therapies). In this setting, the MSM structure may be considered more flexible, with fewer and less restrictive assumptions required versus the PartSA. Researchers should consider important study design features that may influence the generalisability of data when undertaking model conceptualisation.

Cost-effectiveness of home-based cardiac rehabilitation: a systematic review.

OBJECTIVE: Centre-based cardiac rehabilitation (CR) is recognised as cost-effective for individuals following a cardiac event. However, home-based alternatives are becoming increasingly popular, especially since COVID-19, which necessitated alternative modes of care delivery. This review aimed to assess whether home-based CR interventions are cost-effective (vs centre-based CR). METHODS: Using the MEDLINE, Embase and PsycINFO databases, literature searches were conducted in October 2021 to identify full economic evaluations (synthesising costs and effects). Studies were included if they focused on home-based elements of a CR programme or full home-based programmes. Data extraction and critical appraisal were completed using the NHS EED handbook, Consolidated Health Economic Evaluation Reporting Standards and Drummond checklists and were summarised narratively. The protocol was registered on the PROSPERO database (CRD42021286252). RESULTS: Nine studies were included in the review. Interventions were heterogeneous in terms of delivery, components of care and duration. Most studies were economic evaluations within clinical trials (8/9). All studies reported quality-adjusted life years, with the EQ-5D as the most common measure of health status (6/9 studies). Most studies (7/9 studies) concluded that home-based CR (added to or replacing centre-based CR) was cost-effective compared with centre-based options. CONCLUSIONS: Evidence suggests that home-based CR options are cost-effective. The limited size of the evidence base and heterogeneity in methods limits external validity. There were further limitations to the evidence base (eg, limited sample sizes) that increase uncertainty. Future research is needed to cover a greater range of home-based designs, including home-based options for psychological care, with greater sample sizes and the potential to acknowledge patient heterogeneity.

A framework for implementing Patient and Public Involvement in mental health research: The PATHWAY research programme benchmarked against NIHR standards.

BACKGROUND: Patient and Public Involvement (PPI) in research has become a key component recommended by research commissioners, grant award bodies and specified in government policies. Despite the increased call for PPI, few studies have demonstrated how to implement PPI within large-scale research studies. OBJECTIVE: The aim of the current study was to provide a case example of the implementation of a patient advisory group in a large-scale mental health research programme (PATHWAY) and to benchmark this against UK standards. METHOD: A PPI group was incorporated throughout the PATHWAY research programme, from grant development to dissemination. The group attended regular meetings and supported participant recruitment, evaluated patient-facing documents, supported the piloting of the research intervention and co-developed the dissemination and impact strategy. The implementation of PPI throughout the project was benchmarked against the UK standards for PPI. RESULTS: The inclusion of PPI in the PATHWAY project provided tangible changes to the research project (i.e., improving study documents, co-developing dissemination materials) but also proved to be a beneficial experience to PPI members through the development of new skills and the opportunity to provide a patient voice in research. We show how PPI was involved across seven study phases and provide examples of implementation of the six UK standards. The study did not include PPI in data analysis but met all the UK standards for PPI. Challenges regarding practical components (i.e., meeting frequency, language use), increasing diversity and PPI members' knowledge of research were highlighted as areas for further improvement. CONCLUSIONS: We provide a case example of how PPI can be implemented throughout a research lifecycle and we note the barriers faced and make suggestions for PPI in future implementation and research. PATIENT AND PUBLIC CONTRIBUTION: PPI members were involved throughout the lifecycle of the research programme. The PPI lead was a co-author on the manuscript and contributed to report writing.

Discrete choice experiment to investigate preferences for psychological intervention in cardiac rehabilitation.

OBJECTIVE: Cardiac rehabilitation (CR) is offered to people who recently experienced a cardiac event, and often comprises of exercise, education and psychological care. This stated preference study aimed to investigate preferences for attributes of a psychological therapy intervention in CR. METHODS: A discrete choice experiment (DCE) was conducted and recruited a general population sample and a trial sample. DCE attributes included the modality (group or individual), healthcare professional providing care, information provided prior to therapy, location and the cost to the National Health Service (NHS). Participants were asked to choose between two hypothetical designs of therapy, with a separate opt-out included. A mixed logit model was used to analyse preferences. Cost to the NHS was used to estimate willingness to pay (WTP) for aspects of the intervention design. RESULTS: Three hundred and four participants completed the DCE (general public sample (n=262, mean age 47, 48% female) and trial sample (n=42, mean age 66, 45% female)). A preference for receiving psychological therapy was demonstrated by both samples (general population WTP £1081; 95% CI £957 to £1206). The general population appeared to favour individual therapy (WTP £213; 95% CI £160 to £266), delivered by a CR professional (WTP £48; 9% % CI £4 to £93) and with a lower cost (ß=-0.002; p<0.001). Participants preferred to avoid options where no information was received prior to starting therapy (WTP -£106; 95% CI -£153 to -£59). Results for the location attribute were variable and challenging to interpret. CONCLUSIONS: The study demonstrates a preference for psychological therapy as part of a programme of CR, as participants were more likely to opt-in to therapy. Results indicate that some aspects of the delivery which may be important to participants can be tailored to design a psychological therapy. Preference heterogeneity is an issue which may prevent a 'one-size-fits-all' approach to psychological therapy in CR.

Development of a cost of illness inventory questionnaire for children with autism spectrum disorder in South Asia.

BACKGROUND: The economic burden of autism is substantial and includes a range of costs, including healthcare, education, productivity losses, informal care and respite care, among others. In India, approximately, 2 million children aged 2-9 years have autism. Given the likely substantial burden of illness and the need to identify effective and cost-effective interventions, this research aimed to produce a comprehensive cost of illness inventory (COII) suitable for children with autism in South Asia (India) to support future research. METHODS: A structured and iterative design process was followed to create the COII, including literature reviews, interviews with caregivers, pilot testing and translation. Across the development of the COII, thirty-two families were involved in the design and piloting of the tool. The COII was forward translated (from English to Hindi) and back translated. Each stage of the process of development of the COII resulted in the further refinement of the tool. RESULTS: Domains covered in the final COII include education, childcare, relocation, healthcare contacts (outpatient, inpatient, medical emergencies, investigations and medication), religious retreats and rituals, specialist equipment, workshops and training, special diet, support and care, certification, occupational adjustments and government rebates/schemes. Administration and completion of the COII determined it to be feasible to complete in 35 minutes by qualified and trained researchers. The final COII is hosted by REDCap Cloud and is a bilingual instrument (Hindi and English). CONCLUSIONS: The COII was developed using experiences gathered from an iterative process in a metropolitan area within the context of one low- and middle-income country (LMIC) setting, India. Compared to COII tools used for children with autism in high-income country settings, additional domains were required, such as complimentary medication (e.g. religious retreats and homeopathy). The COII will allow future research to quantify the cost of illness of autism in India from a broad perspective and will support relevant economic evaluations. Understanding the process of developing the questionnaire will help researchers working in LMICs needing to adapt the current COII or developing similar questionnaires.

Youth Metacognitive Therapy (YoMeta): protocol for a single-blind randomised feasibility trial of a transdiagnostic intervention versus treatment as usual in 11-16-year-olds with common mental health problems.

BACKGROUND: Mental health disorders in children and young people (CYP) are increasing but the provision of current evidence-based treatment for common mental health problems is limited. Treatment effects vary widely with no clear superiority of a single treatment approach. Further evaluation of contemporary and effective treatments in CYP is needed. Metacognitive therapy (MCT) has shown enhanced efficacy over 'gold standard' approaches in adult mental health, but so far has not been evaluated in a randomised trial of CYP. As such, we aim to assess the acceptability and feasibility of group-MCT for CYP with common mental health problems in comparison to usual treatment within Child and Adolescent Mental Health Services (CAMHS). METHOD: YoMeta is a multicentre, two-arm, single-blind randomised feasibility trial comparing group-MCT to usual care in CYP with common mental health problems in CAMHS. CYP (target sample n = 100) with a common mental health problem will be recruited across at least three CAMHS services in the UK. Participants in the intervention arm will receive up to eight sessions of group-MCT delivered by a CAMHS mental health practitioner. The control arm will receive usual care in CAMHS which includes individual or group-based therapy. Feasibility will be assessed by the success of recruitment, retention, and data quality. Acceptability of the intervention will be assessed by the number of sessions attended and through qualitative interviews aimed at exploring CYP acceptability and understanding of the intervention. Symptoms of psychological distress will be assessed using the Revised Children Anxiety and Depression Scale (RCADS) at 20 weeks. We will also assess psychological well-being, symptoms of depression, metacognitive beliefs, quality of life, and measures to support economic evaluation (health status and health and social care use). Qualitative interviews will be conducted to understand practitioner's views on training and delivery of group-MCT. DISCUSSION: The trial is designed to evaluate the acceptability and feasibility of group-MCT for CYP with common mental health problems. Group-MCT may aid in improving access to treatment, reduce waiting times, and improve outcomes for CYP with common mental health disorders. The study will provide important information and data to evaluate future research potential and confirm sample size estimation for a definitive large-scale RCT to test the effectiveness and cost-effectiveness of group-MCT in CYP. TRIAL REGISTRATION: NCT05260060; ISCTRN18335255.

REACH-ASD: a UK randomised controlled trial of a new post-diagnostic psycho-education and acceptance and commitment therapy programme against treatment-as-usual for improving the mental health and adjustment of caregivers of children recently diagnosed with autism spectrum disorder.

BACKGROUND: Autism is a neurodevelopmental disability affecting over 1% of UK children. The period following a child's autism diagnosis can present real challenges in adaptation for families. Twenty to 50% of caregivers show clinically significant levels of mental health need within the post-diagnostic period and on an ongoing basis. Best practice guidelines recommend timely post-diagnostic family support. Current provision is patchy, largely unevidenced, and a source of dissatisfaction for both families and professionals. There is a pressing need for an evidenced programme of post-diagnostic support focusing on caregiver mental health and adjustment, alongside autism psycho-education. This trial tests the clinical and cost-effectiveness of a new brief manualised psychosocial intervention designed to address this gap. METHODS: This is a multi-centre two-parallel-group single (researcher)-blinded randomised controlled trial of the Empower-Autism programme plus treatment-as-usual versus usual local post-diagnostic offer plus treatment-as-usual. Caregivers of children aged 2-15 years with a recent autism diagnosis will be recruited from North West England NHS or local authority centres. Randomisation is individually by child, with one "index" caregiver per child, stratified by centre, using 2:1 randomisation ratio to assist recruitment and timely intervention. Empower-Autism is a group-based, manualised, post-diagnostic programme that combines autism psycho-education and psychotherapeutic components based on Acceptance and Commitment Therapy to support caregiver mental health, stress management and adjustment to their child's diagnosis. The comparator is any usual local group-based post-diagnostic psycho-education offer. Receipt of services will be specified through health economic data. PRIMARY OUTCOME: caregiver mental health (General Health Questionnaire-30) at 52-week follow-up. SECONDARY OUTCOMES: key caregiver measures (wellbeing, self-efficacy, adjustment, autism knowledge) at 12-, 26- and 52-week follow-up and family and child outcomes (wellbeing and functioning) at 52-week endpoint. SAMPLE: N=380 (approximately 253 intervention/127 treatment-as-usual). Primary analysis will follow intention-to-treat principles using linear mixed models with random intercepts for group membership and repeated measures. Cost-effectiveness acceptability analyses will be over 52 weeks, with decision modelling to extrapolate to longer time periods. DISCUSSION: If effective, this new approach will fill a key gap in the provision of evidence-based care pathways for autistic children and their families. TRIAL REGISTRATION: ISRCTN 45412843 . Prospectively registered on 11 September 2019.

Evaluating Metacognitive Therapy to Improve Treatment of Anxiety and Depression in Cardiovascular Disease: The NIHR Funded PATHWAY Research Programme.

Background: Anxiety and depression contribute to poorer physical and mental health outcomes in cardiac patients. Psychological treatments are not routinely offered in cardiac care and have mixed and small effects. We conducted a series of studies under the PATHWAY research programme aimed at understanding and improving mental health outcomes for patients undergoing cardiac rehabilitation (CR) through provision of metacognitive therapy (MCT). Methods: PATHWAY was a series of feasibility trials, single-blind, multicenter, randomized controlled trials (RCTs), qualitative, stated preferences for therapy and health economics studies. Findings: Patients felt their psychological needs were not met in CR and their narratives of distress could be parsimoniously explained by the metacognitive model. Patients reported they would prefer therapy over no therapy as part of CR, which included delivery by a cardiac professional. Two feasibility studies demonstrated that RCTs of group-based and self-help MCT were acceptable, could be embedded in CR services, and that RCTs of these interventions were feasible. A definitive RCT of group-MCT within CR (n = 332) demonstrated significantly greater reductions in the severity of anxiety and depression, exceeding CR alone, with gains maintained at 12 month follow-up (SMD HADS total score = 0.52 at 4 months and 0.33 at 12 months). A definitive trial of self-help MCT is ongoing. Conclusion: There is a need to better meet the psychological needs of CR patients. Embedding MCT into CR demonstrated high acceptability and improved efficacy on psychological outcomes. Results support roll-out of MCT in CR with evaluation of national implementation. Registration: URL: NCT02420431; ISRCTN74643496; NCT03129282.