Cost-effectiveness of acceptance and commitment therapy for people living with motor neuron disease, and their health-related quality of life.

BACKGROUND: Given the degenerative nature of the condition, people living with motor neuron disease (MND) experience high levels of psychological distress. The purpose of this research was to investigate the cost-effectiveness of acceptance and commitment therapy (ACT), adapted for the specific needs of this population, for improving quality of life. METHODS: A trial-based cost-utility analysis over a 9-month period was conducted comparing ACT plus usual care (n = 97) versus usual care alone (n = 94) from the perspective of the National Health Service. In the primary analysis, quality-adjusted life years (QALYs) were computed using health utilities generated from the EQ-5D-5L questionnaire. Sensitivity analyses and subgroup analyses were also carried out. RESULTS: Difference in costs was statistically significant between the two arms, driven mainly by the intervention costs. Effects measured by EQ-5D-5L were not statistically significantly different between the two arms. The incremental cost-effectiveness was above the £20,000 to £30,000 per QALY gained threshold used in the UK. However, the difference in effects was statistically significant when measured by the McGill Quality of Life-Revised (MQOL-R) questionnaire. The intervention was cost-effective in a subgroup experiencing medium deterioration in motor neuron symptoms. CONCLUSIONS: Despite the intervention being cost-ineffective in the primary analysis, the significant difference in the effects measured by MQOL-R, the low costs of the intervention, the results in the subgroup analysis, and the fact that ACT was shown to improve the quality of life for people living with MND, suggest that ACT could be incorporated into MND clinical services.

Mapping Short Warwick and Edinburgh Mental Wellbeing Scale (SWEMWBS) to Recovering Quality of Life (ReQoL) to estimate health utilities.

BACKGROUND: The Short Warwick and Edinburgh Mental Wellbeing Scale (SWEMWBS) is a widely used non-preference-based measure of mental health in the UK. The primary aim of this paper is to construct an algorithm to translate the SWEMWBS scores to utilities using the Recovering Quality of Life Utility Index (ReQoL-UI) measure. METHODS: Service users experiencing mental health difficulties were recruited in two separate cross-sectional studies in the UK. The following direct mapping functions were used: Ordinary Least Square, Tobit, Generalised Linear Models. Indirect (response) mapping was performed using seemingly unrelated ordered probit to predict responses to each of the ReQoL-UI items and subsequently to predict using UK tariffs of the ReQoL-UI from SWEMWBS. The performance of all models was assessed by the mean absolute errors, root mean square errors between the predicted and observed utilities and graphical representations across the SWEMWBS score range. RESULTS: Analyses were based on 2573 respondents who had complete data on the ReQoL-UI items, SWEMWBS items, age and sex. The direct mapping methods predicted ReQoL-UI scores across the range of SWEMWBS scores reasonably well. Very little differences were found among the three regression specifications in terms of model fit and visual inspection when comparing modelled and actual utility values across the score range of the SWEMWBS. However, when running simulations to consider uncertainty, it is clear that response mapping is superior. CONCLUSIONS: This study presents mapping algorithms from SWEMWBS to ReQoL as an alternative way to generate utilities from SWEMWBS. The algorithm from the indirect mapping is recommended to predict utilities from the SWEMWBS.

A Discrete Choice Experiment to Elicit General Population Preferences Around the Factors Influencing the Choice to Make Clinical Negligence Claims.

OBJECTIVES: This article determines public stated preferences around different factors that influence the choice to make clinical negligence claims against a national healthcare system. METHODS: A large online survey was conducted using a discrete choice experiment (DCE) with the UK general population (N = 1013). DCE tasks involved a single profile and participants chose whether to make a claim for compensation (yes/no) after one of 3 randomly allocated patient safety incident (PSI) "scenarios" of different severities (mild, moderate, severe). DCE attributes described the actions of the healthcare system after a PSI and characteristics of the clinical negligence claims process. The data were modeled separately for each scenario (mild, moderate, severe) using logistic regression. Marginal effects and the probability of making a claim in a baseline case were estimated. RESULTS: Probability of choosing to claim was reduced by receipt of an apology, investigation and prevention of recurrence of the PSI, and longer time until claim decision and increased by an easy and straightforward claims process and high chance of compensation and for the mild scenario higher compensation amounts. Marginal effects and baseline case probabilities differed by scenario severity. CONCLUSIONS: The results suggest the actions of the healthcare system after a PSI and characteristics of the claims process have a larger impact on the probability of making a claim for milder PSIs. For more severe PSIs, a larger probability of making a claim was observed, and the choice was less influenced by the actions of the healthcare system after the PSI and characteristics of the claims process.

Convergent validity of EQ-5D with core outcomes in dementia: a systematic review.

OBJECTIVES: To explore through a systematic review, the convergent validity of EQ-5D (EQ-5D-3L and EQ-5D-5L (total score and dimensions)) with core outcomes in dementia and investigate how this may be impacted by rater-type; with the aim of informing researchers when choosing measures to use in dementia trials. METHODS: To identify articles relevant to the convergent validity of EQ-5D with core dementia outcomes, three databases were electronically searched to September 2022. Studies were considered eligible for inclusion within the review if they included individual level data from people with dementia of any type, collected self and/or proxy reported EQ-5D and collected at least one core dementia outcome measure. Relevant data such as study sample size, stage of dementia and administration of EQ-5D was extracted, and a narrative synthesis was adopted. RESULTS: The search strategy retrieved 271 unique records, of which 30 met the inclusion criteria for the review. Twelve different core outcome measures were used to capture dementia outcomes: cognition, function, and behaviour/mood across the studies. Most studies used EQ-5D-3L (n = 27). Evidence related to the relationship between EQ-5D and measures of function and behaviour/mood was the most robust, with unanimous directions of associations, and more statistically significant findings. EQ-5D dimensions exhibited associations with corresponding clinical outcomes, whereby relationships were stronger with proxy-EQ-5D (than self-report). CONCLUSION: Measuring health-rated quality of life in dementia populations is a complex issue, particularly when considering balancing the challenges associated with both self and proxy report. Published evidence indicates that EQ-5D shows evidence of convergent validity with the key dementia outcomes, therefore capturing these relevant dementia outcomes. The degree of associations with clinical measures was stronger when considering proxy-reported EQ-5D and differed by EQ-5D dimension type. This review has revealed that, despite the limited targeted psychometric evidence pool and reliance on clinical and observational studies, EQ-5D exhibits convergent validity with other dementia outcome measures.

Assessing the psychometric performance of EQ-5D-5L in dementia: a systematic review.

BACKGROUND: EQ-5D is widely used for valuing changes in quality of life for economic evaluation of interventions for people with dementia. There are concerns about EQ-5D-3L in terms of content validity, poor inter-rater agreement and reliability in the presence of cognitive impairment, but there is also evidence to support its use with this population. An evidence gap remains regarding the psychometric properties of EQ-5D-5L. OBJECTIVES: To report psychometric evidence around EQ-5D-5L in people with dementia. METHODS: A systematic review identified primary studies reporting psychometric properties of EQ-5D-5L in people with dementia. Searches were completed up to November 2020. Study selection, data extraction and quality assessment were undertaken independently by at least 2 researchers. RESULTS: Evidence was extracted from 20 articles from 14 unique studies covering a range of dementia severity. Evidence of known group validity from 5 of 7 studies indicated that EQ-5D-5L distinguishes severity of disease measured by cognitive impairment, depression, level of dependence and pain. Convergent validity (9 studies) showed statistically significant correlations of weak and moderate strengths, between EQ-5D-5L scores and scores on other key measures. Statistically significant change was observed in only one of 6 papers that allowed this property to be examined. All seven studies showed a lack of inter-rater reliability between self and proxy reports with the former reporting higher EQ-5D-5L scores than those provided by proxies. Five of ten studies found EQ-5D-5L to be acceptable, assessed by whether the measure could be completed by the PwD and/or by the amount of missing data. As dementia severity increased, the feasibility of self-completing EQ-5D-5L decreased. Three papers reported on ceiling effects, two found some evidence in support of ceiling effects, and one did not. CONCLUSIONS: EQ-5D-5L seems to capture the health of people with dementia on the basis of known-group validity and convergent validity, but evidence is inconclusive regarding the responsiveness of EQ-5D-5L. As disease progresses, the ability to self-complete EQ-5D-5L is diminished.

Estimating a Preference-Based Index for Mental Health From the Recovering Quality of Life Measure: Valuation of Recovering Quality of Life Utility Index.

BACKGROUND: There are increasing concerns about the appropriateness of generic preference-based measures to capture health benefits in the area of mental health. OBJECTIVES: The aim of this study is to estimate preference weights for a new measure, Recovering Quality of Life (ReQoL-10), to better capture the benefits of mental healthcare. METHODS: Psychometric analyses of a larger sample of mental health service users (n = 4266) using confirmatory factor analyses and item response theory were used to derive a health state classification system and inform the selection of health states for utility assessment. A valuation survey with members of the UK public representative in terms of age, sex, and region was conducted using face-to-face interviewer administered time-trade-off with props. A series of regression models were fitted to the data and the best performing model selected for the scoring algorithm. RESULTS: The ReQoL-Utility Index (UI) classification system comprises 6 mental health items and 1 physical health item. Sixty-four health states were valued by 305 participants. The preferred model was a random effects model, with significant and consistent coefficients and best model fit. Estimated utilities modeled for all health states ranged from -0.195 (state worse than dead) to 1 (best possible state). CONCLUSIONS: The development of the ReQoL-UI is based on a novel application of item response theory methods for generating the classification system and selecting health states for valuation. Conventional time-trade-off was used to elicit utility values that are modeled to enable the generation of QALYs for use in cost-utility analysis of mental health interventions.

A Review of the Psychometric Performance of Selected Child and Adolescent Preference-Based Measures Used to Produce Utilities for Child and Adolescent Health.

OBJECTIVE: This review examined the psychometric performance of 4 generic child- and adolescent-specific preference-based measures that can be used to produce utilities for child and adolescent health. METHODS: A systematic search was undertaken to identify studies reporting the psychometric performance of the Child Health Utility (CHU9D), EQ-5D-Y (3L or 5L), and Health Utilities Index Mark 2 (HUI2) or Mark 3 (HUI3) in children and/or adolescents. Data were extracted to assess known-group validity, convergent validity, responsiveness, reliability, acceptability, and feasibility. Data were extracted separately for the dimensions and utility index where this was reported. RESULTS: The review included 76 studies (CHU9D n = 12, EQ-5D-Y-3L n = 20, HUI2 n = 26,HUI3 n = 43), which varied considerably across conditions and sample size. EQ-5D-Y-3L had the largest amount of evidence of good psychometric performance in proportion to the number of studies examining performance. The majority of the evidence related to EQ-5D-Y-3L was based on dimensions. CHU9D was assessed in fewer studies, but the majority of studies found evidence of good psychometric performance. Evidence for HUI2 and HUI3 was more mixed, but the studies were more limited in sample size and statistical power, which was likely to have affected performance. CONCLUSIONS: The heterogeneity of published studies means that the evidence is based on studies across a range of countries, populations and conditions, using different study designs, different languages, different value sets and different statistical techniques. Evidence for CHU9D in particular is based on a limited number of studies. The findings raise concerns about the comparability of self-report and proxy-report responses to generate utility values for children and adolescents.

An item response theory analysis of an item pool for the recovering quality of life (ReQoL) measure.

PURPOSE: ReQoL-10 and ReQoL-20 have been developed for use as outcome measures with individuals aged 16 and over, experiencing mental health difficulties. This paper reports modelling results from the item response theory (IRT) analyses that were used for item reduction. METHODS: From several stages of preparatory work including focus groups and a previous psychometric survey, a pool of items was developed. After confirming that the ReQoL item pool was sufficiently unidimensional for scoring, IRT model parameters were estimated using Samejima's Graded Response Model (GRM). All 39 mental health items were evaluated with respect to item fit and differential item function regarding age, gender, ethnicity, and diagnosis. Scales were evaluated regarding overall measurement precision and known-groups validity (by care setting type and self-rating of overall mental health). RESULTS: The study recruited 4266 participants with a wide range of mental health diagnoses from multiple settings. The IRT parameters demonstrated excellent coverage of the latent construct with the centres of item information functions ranging from - 0.98 to 0.21 and with discrimination slope parameters from 1.4 to 3.6. We identified only two poorly fitting items and no evidence of differential item functioning of concern. Scales showed excellent measurement precision and known-groups validity. CONCLUSION: The results from the IRT analyses confirm the robust structure properties and internal construct validity of the ReQoL instruments. The strong psychometric evidence generated guided item selection for the final versions of the ReQoL measures.

Public involvement in health outcomes research: lessons learnt from the development of the recovering quality of life (ReQoL) measures.

BACKGROUND: To provide a model for Public involvement (PI) in instrument development and other research based on lessons learnt in the co-production of a recently developed mental health patient reported outcome measure called Recovering Quality of Life (ReQoL). While service users contributed to the project as research participants, this paper focuses on the role of expert service users as research partners, hence referred to as expert service users or PI. METHODS: At every stage of the development, service users influenced the design, content and face validity of the measure, collaborating with other researchers, clinicians and stakeholders who were central to this research. Expert service users were integral to the Scientific Group which was the main decision-making body, and also provided advice through the Expert Service User Group. RESULTS: During the theme and item generation phase (stage 1) expert service users affirmed the appropriateness of the seven domains of the Patient Reported Outcome Measure (activity, hope, belonging and relationships, self-perception, wellbeing, autonomy, and physical health). Expert service users added an extra 58 items to the pool of 180 items and commented on the results from the face and content validity testing (stage 2) of a refined pool of 88. In the item reduction and scale generation phase (stage 3), expert service users contributed to discussions concerning the ordering and clustering of the themes and items and finalised the measures. Expert service users were also involved in the implementation and dissemination of ReQoL (stage 4). Expert service users contributed to the interpretation of findings, provided inputs at every stage of the project and were key decision-makers. The challenges include additional work to make the technical materials accessible, extra time to the project timescales, including time to achieve consensus from different opinions, sometimes strongly held, and extra costs. CONCLUSION: This study demonstrates a successful example of how PI can be embedded in research, namely in instrument development. The rewards of doing so cannot be emphasised enough but there are challenges, albeit surmountable ones. Researchers should anticipate and address those challenges during the planning stage of the project.

Exploring the item sets of the Recovering Quality of Life (ReQoL) measures using factor analysis.

PURPOSE: This paper presents two studies exploring the latent structure of item sets used in the development of the Recovering Quality of Life mental health outcome measures: ReQoL-10 and ReQoL-20. METHOD: In study 1, 2262 participants completed an initial set of 61 items. In study 2, 4266 participants completed a reduced set of 40 items. Study 2 evaluated two formats of the questionnaires: one version where the items were intermingled and one where the positively worded and negatively worded items were presented as two separate blocks. Exploratory and confirmatory factor analyses were conducted on both datasets where models were specified using ordinal treatment of the item responses. Dimensionality based on the conceptual framework and methods effects reflecting the mixture of positively worded and negatively worded items were explored. Factor invariance was tested across the intermingled and block formats. RESULTS: In both studies, a bi-factor model (study 1: RMSEA = 0.061; CFI = 0.954; study 2: RMSEA = 0.066; CFI = 0.971) with one general factor and two local factors (positively worded questions and negatively worded questions) was preferred. The loadings on the general factor were higher than on the two local factors suggesting that the ReQoL scale scores can be understood in terms of a general factor. Insignificant differences were found between the intermingled and block formats. CONCLUSIONS: The analyses confirmed that the ReQoL item sets are sufficiently unidimensional to proceed to item response theory analysis. The model was robust across different ordering of positive and negative items.

Recovering Quality of Life (ReQoL): a new generic self-reported outcome measure for use with people experiencing mental health difficulties.

BACKGROUND: Outcome measures for mental health services need to adopt a service-user recovery focus. Aims To develop and validate a 10- and 20-item self-report recovery-focused quality of life outcome measure named Recovering Quality of Life (ReQoL). METHOD: Qualitative methods for item development and initial testing, and quantitative methods for item reduction and scale construction were used. Data from >6500 service users were factor analysed and item response theory models employed to inform item selection. The measures were tested for reliability, validity and responsiveness. RESULTS: ReQoL-10 and ReQoL-20 contain positively and negatively worded items covering seven themes: activity, hope, belonging and relationships, self-perception, well-being, autonomy, and physical health. Both versions achieved acceptable internal consistency, test-retest reliability (>0.85), known-group differences, convergence with related measures, and were responsive over time (standardised response mean (SRM) > 0.4). They performed marginally better than the Short Warwick-Edinburgh Mental Well-being Scale and markedly better than the EQ-5D. CONCLUSIONS: Both versions are appropriate for measuring service-user recovery-focused quality of life outcomes. Declaration of interest M.B. and J.Co. were members of the research group that developed the Clinical Outcomes in Routine Evaluation (CORE) outcome measures.

The importance of content and face validity in instrument development: lessons learnt from service users when developing the Recovering Quality of Life measure (ReQoL).

PURPOSE: Service user involvement in instrument development is increasingly recognised as important, but is often not done and seldom reported. This has adverse implications for the content validity of a measure. The aim of this paper is to identify the types of items that service users felt were important to be included or excluded from a new Recovering Quality of Life measure for people with mental health difficulties. METHODS: Potential items were presented to service users in face-to-face structured individual interviews and focus groups. The items were primarily taken or adapted from current measures and covered themes identified from earlier qualitative work as being important to quality of life. Content and thematic analysis was undertaken to identify the types of items which were either important or unacceptable to service users. RESULTS: We identified five key themes of the types of items that service users found acceptable or unacceptable; the items should be relevant and meaningful, unambiguous, easy to answer particularly when distressed, do not cause further upset, and be non-judgemental. Importantly, this was from the perspective of the service user. CONCLUSIONS: This research has underlined the importance of service users' views on the acceptability and validity of items for use in developing a new measure. Whether or not service users favoured an item was associated with their ability or intention to respond accurately and honestly to the item which will impact on the validity and sensitivity of the measure.

Sensitivity and responsiveness of the EQ-5D-3L in patients with uncontrolled focal seizures: an analysis of Phase III trials of adjunctive brivaracetam.

PURPOSE: Preference-based measures are required to measure the impact of interventions for cost-effectiveness analysis. This study assessed the psychometric performance of the EQ-5D-3L in adults with uncontrolled focal (partial-onset) seizures. METHODS: Data from three Phase III studies of an antiepileptic drug (adjunctive brivaracetam; n = 1095) were used. Analysis included correlations between EQ-5D-3L and Quality of Life in Epilepsy Inventory (QOLIE-31P) and seizure frequency. Known group validity was based on ability of the EQ-5D-3L to discriminate between baseline QOLIE-31P total scores, seizure type and number of antiepileptic drugs using effect sizes (ES). Responsiveness assessed proportions reporting highest or lowest scores, overall change using standardized response means (SRM) and change by responder and clinician/patient evaluation groups using ES. RESULTS: Correlations were weak to moderate (ρ = 0.2-0.4) between EQ-5D-3L dimensions and QOLIE-31P subscales, apart from medication effects (ρ < 0.1); seizure frequency was not associated with either measure. Known group analysis had small ES. A quarter (24.9%) of patients had a baseline EQ-5D-3L utility score of 1 (full health) but lower average QOLIE-31P scores. SRMs were small (<0.1) in EQ-5D-3L compared with 0.1-0.4 for QOLIE-31P subscales. Results across the studies were mixed for responder status and clinician/patient evaluation of improvement for EQ-5D-3L. CONCLUSIONS: EQ-5D-3L had weak-to-moderate correlations with QOLIE-31P and varied with QOLIE-31P severity groups, but showed less responsiveness than QOLIE-31P. Given this lack of sensitivity, EQ-5D-3L may not be appropriate for measuring the impact of interventions in cost-effectiveness analysis in this population and disease-specific preference-based measures may be more appropriate.

Patient-reported outcome measures in patients with peripheral arterial disease: a systematic review of psychometric properties.

BACKGROUND: Peripheral arterial disease (PAD) is generally associated with considerable morbidity and reduced quality of life. Patient-reported outcome measures (PROMs) provide important information about the burden of disease and impact of treatment in affected patients. OBJECTIVES: The objective of the review was to identify and appraise studies reporting the psychometric evaluation of PROMs administered to a specified population of patients with PAD with a view to recommending suitable PROMs. METHODS: A systematic review of peer-reviewed English language articles was undertaken to identify primary studies reporting psychometric properties of PROMs in English-speaking patients with various stages of PAD. Comprehensive searches were completed up until January 2015. Study selection, data extraction and quality assessment were undertaken independently by at least two researchers. Findings were presented as tabular and narrative summaries based on accepted guidance. RESULTS: Psychometric evaluation of 6 generic and 7 condition-specific PROMs reported in 14 studies contributed data to the review. The frequently reported measure was the SF-36 (n = 11 studies); others included the Walking Impairment Questionnaire (n = 8 studies), EQ-5D (n = 5 studies) and the Vascular Quality of Life Questionnaire (n = 3 studies). Studies included a diverse PAD population and varied in methodology, including approach to validation of PROMs. CONCLUSIONS: Various PROMs have been validated in patients with PAD but no study provided evidence of a full psychometric evaluation in the patient population. Careful selection is required to identify reliable and valid PROMs to use in clinical and research settings.

Quality of life impact of mental health conditions in England: results from the adult psychiatric morbidity surveys.

BACKGROUND: The main objective is to present health state utility estimates for a broad range of mental health conditions including anxiety, depression, long-term depression, obsessive compulsive disorder, phobia, panic disorder, psychosis, alcohol and drug dependency that can be used in economic models. METHODS: This study uses pooled data from the Adult Psychiatric Morbidity Surveys carried out in 2000 and 2007 of a representative sample of the general population in England. Health state utility values measured by the SF-6D and EQ-5D indices are the dependent variables. Independent variables include background characteristics, mental health and physical health conditions. Regression models were estimated using OLS for the SF-6D and tobit for EQ-5D. Further regressions were carried out to consider the impact of mental health and physical health morbidities and the impact of severity of conditions on utility values. RESULTS: Mental health conditions tend to have a larger impact on health state utility values than physical health conditions. The mental health conditions associated with the highest decrements in utility are: depression, mixed anxiety and depressive disorders and long-term depression. Interaction terms used to model the effect of co-morbidities are generally found to be positive implying that simply adding the utility decrements for two mental health conditions overestimates the burden of the disease. CONCLUSIONS: This paper presents reliable and representative community based mean SF-6D and EQ-5D estimates with standard errors for health state utility values across a broad range of mental health conditions that can be used in cost effectiveness modelling.

Are policy decisions on surgical procedures informed by robust economic evidence? A systematic review.

OBJECTIVES: The aim of this study was to examine the empirical and methodological cost-effectiveness evidence of surgical interventions for breast, colorectal, or prostate cancer. METHODS: A systematic search of seven databases including MEDLINE, EMBASE, and NHSEED, research registers, the NICE Web site and conference proceedings was conducted in April 2012. Study quality was assessed in terms of meeting essential, preferred and UK NICE specific requirements for economic evaluations. RESULTS: The seventeen (breast = 3, colorectal = 7, prostate = 7) included studies covered a broad range of settings (nine European; eight non-European) and six were published over 10 years ago. The populations, interventions and comparators were generally well defined. Very few studies were informed by literature reviews and few used synthesized clinical evidence. Although the interventions had potential differential effects on recurrence and mortality rates, some studies used relatively short time horizons. Univariate sensitivity analyses were reported in all studies but less than a third characterized all uncertainty with a probabilistic sensitivity analysis. Although a third of studies incorporated patients' health-related quality of life data, only four studies used social tariff values. CONCLUSIONS: There is a dearth of recent robust evidence describing the cost-effectiveness of surgical interventions in the management of breast, colorectal and prostate cancers. Many of the recent publications did not satisfy essential methodological requirements such as using clinical evidence informed by a systematic review and synthesis. Given the ratio of potential benefit and harms associated with cancer surgery and the volume of resources consumed by these, there is an urgent need to increase economic evaluations of these technologies.

Understanding UK public views on normative decisions made to value health-related quality of life in children: A qualitative study.

Developing methodology for measuring and valuing child health-related quality of life (HRQoL) is a priority for health technology agencies. One aspect of this is normative decisions that are made in child HRQoL valuation. This qualitative study aimed to better understand adult public opinion on the normative questions of whose preferences to elicit (adults, children, or both) and from which perspective (who should be imagined living in impaired health), when valuing child HRQoL. Opinions of the adult UK public (N = 32) were solicited using online semi-structured focus groups, featuring a breadth of age, sex, ethnicities, and responsibility for children under 18 years. Participants were provided with bespoke informational material on health state valuation and were probed for their views. Arguments for and against different positions were discussed. Data was analysed using framework analysis. Participants demonstrated near-to-universal agreement that children should be involved in valuation in some form, yet this should differ depending on age or maturity. There was strong support for approaches combining involvement from children and adults (e.g., their parents), especially for younger children. There was little intuitive support for the 'taxpayer argument' for asking taxpaying adults. In the context of greater involvement of children in valuation, most participants supported using an 'own' perspective. Most participants thought that valuation study participants should know the exercise is about valuing child health states for ethical reasons. Informed views from the UK public on who should be asked and with what perspective when valuing child HRQoL appear to differ from normative positions previously advocated by some health economists, such as prioritising the preferences of taxpaying adults. In contrast, the results suggest including adults and children in valuation, with the proviso that the children are of an appropriate age and level of maturity, and that an own perspective is used wherever possible.

Exploring the Factors that Drive Clinical Negligence Claims: Stated Preferences of Those Who Have Experienced Unintended Harm.

BACKGROUND: Better understanding of the factors that influence patients to make a financial claim for compensation is required to inform policy decisions. This study aimed to assess the relative importance of factors that influence those who have experienced a patient safety incident (PSI) to make a claim for compensation. METHOD: Participants completed an online discrete choice experiment (DCE) involving 10 single profile tasks where they chose whether or not to file a claim. DCE data were modelled using logistic, mixed logit and latent class regressions; scenario analyses, external validity, and willingness to accept were also conducted. RESULTS: A total of 1029 participants in the United Kingdom responded to the survey. An appropriate apology and a satisfactory investigation reduced the likelihood of claiming. Respondents were more likely to claim if they could hold those responsible accountable, if the process was simple and straightforward, if the compensation amount was higher, if the likelihood of compensation was high or uncertain, if the time to receive a decision was quicker, and if they used the government compensation scheme. Men are more likely to claim for low impact PSIs. DISCUSSION AND CONCLUSIONS: The actions taken by the health service after a PSI, and people's perceptions about the probability of success and the size of potential reward, can influence whether a claim is made. Results show the importance of giving an appropriate apology and conducting a satisfactory investigation. This stresses the importance around how patients are treated after a PSI in influencing the clinical negligence claims that are made.

Behaviour change intervention for toothbrushing (lesson and text messages) to prevent dental caries in secondary school pupils: The BRIGHT randomized control trial.

OBJECTIVES: This multicentre, assessor-blinded, two-arm cluster randomized trial evaluated the clinical and cost-effectiveness of a behaviour change intervention promoting toothbrushing for preventing dental caries in UK secondary schools. METHODS: Pupils aged 11-13 years with their own mobile telephone attending secondary schools with above average free school meals eligibility were randomized (at year-group level) to receive a lesson and twice-daily text messages or to usual care. Year-groups (n = 84) from 42 schools including 4680 pupils (intervention, n = 2262; control, n = 2418) were randomized. RESULTS: In 2383 participants with valid data at baseline and 2.5 years, the primary outcome of presence of at least one treated or untreated carious lesion (D4-6 MFT [Decayed, Missing and Filled Teeth] in permanent teeth using International Caries Detection and Assessment System) was 44.6% in the intervention group and 43.0% in control (odds ratio [OR] 1.04, 95% CI 0.85-1.26, p = .72). There were no statistically significant differences in secondary outcomes of presence of at least one treated or untreated carious lesion (D1-6 MFT), number of D4-6 MFT and D1-6 MFT, plaque and bleeding scores or health-related- (Child Health Utility 9D) or oral health-related- quality of life (CARIES-QC). However, twice-daily toothbrushing, reported by 77.6% of pupils at baseline, increased at 6 months (intervention, 86.9%; control, 83.0%; OR 1.30, 95% CI 1.03-1.63, p = .03), but returned to no difference at 2.5 years (intervention, 81.0%; control, 79.9%; OR 1.05, 95% CI 0.84-1.30, p = .69). Estimated incremental costs and quality-adjusted life-years (QALYs) of the intervention, relative to control, were £1.02 (95% CI -1.29 to 3.23) and -0.003 (95% CI -0.009 to 0.002), respectively, with a 7% chance of being cost-effective (£20 000/QALY gained threshold). CONCLUSION: There was no evidence of statistically significant difference for caries prevalence at 2.5-years. The intervention's positive 6-month toothbrushing behaviour change did not translate into caries reduction. (ISRCTN 12139369). COVID-19 pandemic adversly affected follow-up.

Uptake of self-management education programmes for people with type 2 diabetes in primary care through the embedding package: a cluster randomised control trial and ethnographic study.

BACKGROUND: Self-management education programmes are cost-effective in helping people with type 2 diabetes manage their diabetes, but referral and attendance rates are low. This study reports on the effectiveness of the Embedding Package, a programme designed to increase type 2 diabetes self-management programme attendance in primary care. METHODS: Using a cluster randomised design, 66 practices were randomised to: (1) a wait-list group that provided usual care for nine months before receiving the Embedding Package for nine months, or (2) an immediate group that received the Embedding Package for 18 months. 'Embedders' supported practices and self-management programme providers to embed programme referral into routine practice, and an online 'toolkit' contained embedding support resources. Patient-level HbA1c (primary outcome), programme referral and attendance data, and clinical data from 92,977 patients with type 2 diabetes were collected at baseline (months - 3-0), step one (months 1-9), step 2 (months 10-18), and 12 months post-intervention. An integrated ethnographic study including observations, interviews, and document analysis was conducted using interpretive thematic analysis and Normalisation Process Theory. RESULTS: No significant difference was found in HbA1c between intervention and control conditions (adjusted mean difference [95% confidence interval]: -0.10 [-0.38, 0.18] mmol/mol; -0.01 [-0.03, 0.02] %). Statistically but not clinically significantly lower levels of HbA1c were found in people of ethnic minority groups compared with non-ethnic minority groups during the intervention condition (-0.64 [-1.08, -0.20] mmol/mol; -0.06% [-0.10, -0.02], p = 0.004), but not greater self-management programme attendance. Twelve months post-intervention data showed statistically but not clinically significantly lower HbA1c (-0.56 [95% confidence interval: -0.71, -0.42] mmol/mol; -0.05 [-0.06, -0.04] %; p < 0.001), and higher self-management programme attendance (adjusted odds ratio: 1.13; 95% confidence interval: 1.02, 1.25; p = 0.017) during intervention conditions. Themes identified through the ethnographic study included challenges for Embedders in making and sustaining contact with practices and providers, and around practices' interactions with the toolkit. CONCLUSIONS: Barriers to implementing the Embedding Package may have compromised its effectiveness. Statistically but not clinically significantly improved HbA1c among ethnic minority groups and in longer-term follow-up suggest that future research exploring methods of embedding diabetes self-management programmes into routine care is warranted. TRIAL REGISTRATION: ISRCTN23474120, registered 05/04/2018.