Health Care Costs After Genome-Wide Sequencing for Children With Rare Diseases in England and Canada.

Importance: Etiologic diagnoses for rare diseases can involve a diagnostic odyssey, with repeated health care interactions and inconclusive diagnostics. Prior studies reported cost savings associated with genome-wide sequencing (GWS) compared with cytogenetic or molecular testing through rapid genetic diagnosis, but there is limited evidence on whether diagnosis from GWS is associated with reduced health care costs. Objective: To measure changes in health care costs after diagnosis from GWS for Canadian and English children with suspected rare diseases. Design, Setting, and Participants: This cohort study was a quasiexperimental retrospective analysis across 3 distinct English and Canadian cohorts, completed in 2023. Mixed-effects generalized linear regression was used to estimate associations between GWS and costs in the 2 years before and after GWS. Difference-in-differences regression was used to estimate associations of genetic diagnosis and costs. Costs are in 2019 US dollars. GWS was conducted in a research setting (Genomics England 100 000 Genomes Project [100KGP] and Clinical Assessment of the Utility of Sequencing and Evaluation as a Service [CAUSES] Research Clinic) or clinical outpatient setting (publicly reimbursed GWS in British Columbia [BC], Canada). Participants were children with developmental disorders, seizure disorders, or both undergoing GWS between 2014 and 2019. Data were analyzed from April 2021 to September 2023. Exposures: GWS and genetic diagnosis. Main Outcomes and Measures: Annual health care costs and diagnostic costs per child. Results: Study cohorts included 7775 patients in 100KGP, among whom 788 children had epilepsy (mean [SD] age at GWS, 11.6 [11.1] years; 400 female [50.8%]) and 6987 children had an intellectual disability (mean [SD] age at GWS, 8.2 [8.4] years; 2750 female [39.4%]); 77 patients in CAUSES (mean [SD] age at GWS, 8.5 [4.4] years; 33 female [42.9%]); and 118 publicly reimbursed GWS recipients from BC (mean [SD] age at GWS, 5.5 [5.2] years; 58 female [49.2%]). GWS diagnostic yield was 143 children (18.1%) for those with epilepsy and 1323 children (18.9%) for those with an intellectual disability in 100KGP, 47 children (39.8%) in the BC publicly reimbursed setting, and 42 children (54.5%) in CAUSES. Mean annual per-patient spending over the study period was $5283 (95% CI, $5121-$5427) for epilepsy and $3373 (95% CI, $3322-$3424) for intellectual disability in the 100KGP, $724 (95% CI, $563-$886) in CAUSES, and $1573 (95% CI, $1372-$1773) in the BC reimbursed setting. Receiving a genetic diagnosis from GWS was not associated with changed costs in any cohort. Conclusions and Relevance: In this study, receiving a genetic diagnosis was not associated with cost savings. This finding suggests that patient benefit and cost-effectiveness should instead drive GWS implementation.

Association between health insurance cost-sharing and choice of hospital tier for cardiovascular diseases in China: a prospective cohort study.

Background: Hospitals in China are classified into tiers (1, 2 or 3), with the largest (tier 3) having more equipment and specialist staff. Differential health insurance cost-sharing by hospital tier (lower deductibles and higher reimbursement rates in lower tiers) was introduced to reduce overcrowding in higher tier hospitals, promote use of lower tier hospitals, and limit escalating healthcare costs. However, little is known about the effects of differential cost-sharing in health insurance schemes on choice of hospital tiers. Methods: In a 9-year follow-up of a prospective study of 0.5 M adults from 10 areas in China, we examined the associations between differential health insurance cost-sharing and choice of hospital tiers for patients with a first hospitalisation for stroke or ischaemic heart disease (IHD) in 2009-2017. Analyses were performed separately in urban areas (stroke: n = 20,302; IHD: n = 19,283) and rural areas (stroke: n = 21,130; IHD: n = 17,890), using conditional logit models and adjusting for individual socioeconomic and health characteristics. Findings: About 64-68% of stroke and IHD cases in urban areas and 27-29% in rural areas chose tier 3 hospitals. In urban areas, higher reimbursement rates in each tier and lower tier 3 deductibles were associated with a greater likelihood of choosing their respective hospital tiers. In rural areas, the effects of cost-sharing were modest, suggesting a greater contribution of other factors. Higher socioeconomic status and greater disease severity were associated with a greater likelihood of seeking care in higher tier hospitals in urban and rural areas. Interpretation: Patient choice of hospital tiers for treatment of stroke and IHD in China was influenced by differential cost-sharing in urban areas, but not in rural areas. Further strategies are required to incentivise appropriate health seeking behaviour and promote more efficient hospital use. Funding: Wellcome Trust, Medical Research Council, British Heart Foundation, Cancer Research UK, Kadoorie Charitable Foundation, China Ministry of Science and Technology, and National Natural Science Foundation of China.

Identifying Preferred Features of Weight Loss Programs for Adults With or at Risk of Type 2 Diabetes: A Discrete Choice Experiment With 3,960 Adults in the U.K.

OBJECTIVE: To understand preferences for features of weight loss programs among adults with or at risk of type 2 diabetes in the U.K. RESEARCH DESIGN AND METHODS: We conducted a discrete choice experiment with 3,960 U.K. adults living with overweight (n = 675 with type 2 diabetes). Preferences for seven characteristics of weight loss programs were analyzed. Simulations from choice models using the experimental data predicted uptake of available weight loss programs. Patient groups comprising those who have experience with weight loss programs, including from minority communities, informed the experimental design. RESULTS: Preferences did not differ between individuals with and without type 2 diabetes. Preferences were strongest for type of diet. Healthy eating was most preferred relative to total diet replacement (odds ratio [OR] 2.24; 95% CI 2.04-2.44). Individual interventions were more popular than group interventions (OR 1.40; 95% CI 1.34-1.47). Participants preferred programs offering weight loss of 10-15 kg (OR 1.37; 95% CI 1.28-1.47) to those offering loss of 2-4 kg. Online content was preferred over in-person contact (OR 1.24; 95% CI 1.18-1.30). There were few differences in preferences by gender or ethnicity, although weight loss was more important to women than to men, and individuals from ethnic minority populations identified more with programs where others shared their characteristics. Modeling suggested that tailoring programs to individual preferences could increase participation by ∼17 percentage points (68% in relative terms). CONCLUSIONS: Offering a range of weight loss programs targeting the preferred attributes of different patient groups could potentially encourage more people to participate in weight loss programs and support those living with overweight to reduce their weight.

Defining treatment success in children with surgical conditions.

OBJECTIVES: Develop a score summarising how successfully a child with any surgical condition has been treated, and test the clinical validity of the score. DESIGN: Discrete choice experiment (DCE), and secondary analysis of data from six UK-wide prospective cohort studies. PARTICIPANTS: 253 people with lived experience of childhood surgical conditions, 114 health professionals caring for children with surgical conditions and 753 members of the general population completed the DCE. Data from 1383 children with surgical conditions were used in the secondary analysis. MAIN OUTCOME MEASURES: Normalised importance value of attribute (NIVA) for number/type of operations, hospital-treated infections, quality of life and duration of survival (reference attribute). RESULTS: Quality of life and duration of survival were the most important attributes in deciding whether a child had been successfully treated. Parents, carers and previously treated adults placed equal weight on both attributes (NIVA=0.996; 0.798 to 1.194). Healthcare professionals placed more weight on quality of life (NIVA=1.469; 0.950 to 1.987). The general population placed more weight on survival (NIVA=0.823; 95% CI 0.708 to 0.938). The resulting score (the Children's Surgery Outcome Reporting (CSOR) Treatment Success Score (TSS)) has the best possible value of 1, a value of 0 describes palliation and values less than 0 describe outcomes worse than palliation. CSOR TSSs varied clinically appropriately for infants whose data were included in the UK-wide cohort studies. CONCLUSIONS: The CSOR TSS summarises how successfully children with surgical conditions have been treated, and can therefore be used to compare hospitals' observed and expected outcomes.

Responses to Real-World and Hypothetical Menthol Flavor Bans Among US Young Adults Who Smoke Menthol Cigarettes.

INTRODUCTION: Menthol cigarette bans have been implemented in some US states and localities, and a federal ban is being proposed by the FDA. This study asks how young adults who use menthol cigarettes respond to changes in menthol cigarette availability. AIMS AND METHODS: An online survey of young adults ages 18-34 who reported smoking menthol cigarettes on ≥7 of 30 days around Thanksgiving 2019 (n = 734), oversampling Massachusetts-the first state with a menthol ban. Participants reported their tobacco use behavior following real-world menthol cigarette bans or predicted their behavior under a hypothetical federal ban. RESULTS: Most respondents who exclusively smoked versus dual used with e-cigarettes continued smoking/using combustible tobacco following real-world bans (95.3% vs. 86.9%), accessing menthol cigarettes from other jurisdictions. Fewer who smoked exclusively responded by using e-cigarettes compared to those who dual used (3.9% vs. 43.7%). Quitting all tobacco use (ie, no smoking, vaping, or any tobacco use) was uncommon for both groups (3.6% vs. 9.0%). Under a hypothetical ban, majorities of those who exclusively smoke and who dual use predicted they would continue smoking (72.2% vs. 71.8%); fewer who smoke exclusively would use e-cigarettes compared to those who dual use (14.7% vs. 41.4%). Those who smoke exclusively were more likely to report quitting all tobacco compared to those who dual use (29.6% vs. 12.4%). CONCLUSIONS: Under real-world and hypothetical menthol cigarette bans, most respondents continued smoking. However, more young adults continued smoking following real-world bans, reflecting the limitations of local/state restrictions when menthol cigarettes are available in other jurisdictions. IMPLICATIONS: This survey asked young adults who use menthol cigarettes how they responded to real-world changes in the availability of menthol cigarettes; 89% reported continuing to smoke. Those who smoked exclusively were far less likely to respond by switching to e-cigarettes compared to people who dual used both products. Under a hypothetical federal menthol cigarette ban, 72% of young adults predicted that they would continue smoking. Quitting all tobacco was less common in the real-world scenario compared to the hypothetical ban. Access to menthol cigarettes in other jurisdictions and flavored cigars likely dampen the public health benefit of menthol cigarette bans.

Responses to real-world and hypothetical e-cigarette flavor bans among US young adults who use flavored e-cigarettes.

INTRODUCTION: E-cigarette flavor bans could reduce or exacerbate population health harms. To determine how US e-cigarette flavor restrictions might influence tobacco use behavior, this study assesses responses to real-world and hypothetical flavor bans among young adults who use flavored e-cigarettes. METHODS: An online, national survey of young adults ages 18-34 who use flavored e-cigarettes was conducted in 2021 (n=1,253), oversampling states affected by e-cigarette flavor restrictions. Participants were asked about their responses to real-world changes in the availability of flavored e-cigarettes. Unaffected participants were asked to predict their responses under a hypothetical federal e-cigarette flavor ban. RESULTS: The most common response to real-world changes in flavored e-cigarettes availability was to continue vaping (~80%). Among those who exclusively vaped, 12.5% switched to combustible tobacco. Quitting all forms of tobacco was selected by 5.3% of those exclusively vape vs. 4.2% who dual use. Under a hypothetical federal ban, more than half of respondents stated they would continue vaping; 20.9% and 42.5% of those who exclusively vape vs dual use would use combustible tobacco. Quitting all tobacco products was endorsed by 34.5% and 17.2% of those who exclusively vape vs dual use. CONCLUSIONS: Young adults who vape flavored e-cigarettes have mixed responses to e-cigarette flavor bans. Under both real-world and hypothetical e-cigarette flavor bans, most who use flavored e-cigarettes continue vaping. Under a real-world ban, the second most common response among those who exclusively vape is to switch to smoking; under a hypothetical federal ban, it is to quit all tobacco. IMPLICATIONS: This is the first national survey to directly ask young adults who use flavored e-cigarettes about their responses to real-world changes in flavored e-cigarette availability due to state and local flavor restrictions. The survey also asked individuals to predict their responses under a hypothetical federal e-cigarette flavor ban. Most who use flavored e-cigarettes would continue vaping following e-cig flavor restrictions, but many would switch to or continue using combustible tobacco, highlighting potential negative public health consequences of these policies. Policymakers must consider the impact of e-cigarette flavor bans on both e-cigarette and cigarette use.

Trends in all-cause mortality among adults with diagnosed type 2 diabetes in West Malaysia: 2010 - 2019.

AIMS: We determined 10-year all-cause mortality trends in diagnosed type 2 diabetes (T2D) population in West Malaysia, a middle-income country in the Western-Pacific region. METHODS: One million T2D people aged 40-79 registered in the National Diabetes Registry (2009-2018) were linked to death records (censored on 31 December 2019). Standardized absolute mortality rates and standardized mortality ratios (SMRs) were estimated relative to the Malaysian general population, and standardized to the 2019 registry population with respect to sex, age group, and disease duration. RESULTS: Overall all-cause standardized mortality rates were unchanged in both sexes. Rates increased in males aged 40-49 (annual average percent change [AAPC]: 2.46 % [95 % CI 0.42 %, 4.55 %]) and 50-59 (AAPC: 1.91 % [95 % CI 0.73 %, 3.10 %]), and females aged 40-49 (AAPC: 3.39 % [95 % CI 1.32 %, 5.50 %]). In both sexes, rates increased among those with 1) > 15 years disease duration, 2) prior cardiovascular disease, and 3) Bumiputera (Malay/native) ethnicity. The overall SMR was 1.83 (95 % CI 1.80, 1.86) for males and 1.85 (95 % CI 1.82, 1.89) for females, being higher in younger age groups and showed an increasing trend in those with either > 15 years disease duration or prior cardiovascular disease. CONCLUSIONS: Mortality trends worsened in certain T2D population in Malaysia.

Derivation of a UK preference-based value set for the Short Warwick-Edinburgh Mental Well-being Scale (SWEMWBS) to allow estimation of Mental Well-being Adjusted Life Years (MWALYs).

BACKGROUND: The Mental Well-being Adjusted Life Year (MWALY) is an alternative outcome measure to the quality-adjusted life year (QALY) in economic evaluations of interventions aimed at improving mental well-being. However, there is a lack of preference-based mental well-being instruments for capturing population mental well-being preferences. OBJECTIVES: To derive a UK preference-based value set for the Short Warwick-Edinburgh Mental Well-being Scale (SWEMWBS). METHODS: 225 participants that were interviewed between December 2020 and August 2021 completed 10 composite time trade-off (C-TTO) and 10 discrete choice experiment (DCE) interviewer-administered exercises. Heteroskedastic Tobit and conditional logit models were used to model C-TTO and DCE responses respectively. The DCE utility values were rescaled to a C-TTO comparable scale through anchoring and mapping. An inverse variance weighting hybrid model (IVWHM) was used to derive weighted-average coefficients from the modelled C-TTO and DCE coefficients. Model performance was assessed using statistical diagnostics. RESULTS: The valuation responses confirmed the feasibility and face validity of the C-TTO and DCE techniques. Apart from the main effects models, statistically significant associations were estimated between the predicted C-TTO value and participants' SWEMWBS scores, gender, ethnicities, education levels, and the interaction terms between age and useful feeling. The IVWHM was the most optimal model with the fewest logically inconsistent coefficients and the lowest pooled standard errors. The utility values generated by the rescaled DCE models and the IVWHM were generally higher than those of the C-TTO model. The predictive ability of the two DCE rescaling methods was similar according to the mean absolute deviation and root mean square deviation statistics. CONCLUSIONS: This study has produced the first preference-based value set for a measure of mental well-being. The IVWHM provided a desirable blend of both C-TTO and DCE models. The value set derived by this hybrid approach can be used for cost-utility analyses of mental well-being interventions.

COVID-19 vaccination, risk-compensatory behaviours, and contacts in the UK.

The physiological effects of vaccination against SARS-CoV-2 (COVID-19) are well documented, yet the behavioural effects not well known. Risk compensation suggests that gains in personal safety, as a result of vaccination, are offset by increases in risky behaviour, such as socialising, commuting and working outside the home. This is potentially important because transmission of SARS-CoV-2 is driven by contacts, which could be amplified by vaccine-related risk compensation. Here, we show that behaviours were overall unrelated to personal vaccination, but-adjusting for variation in mitigation policies-were responsive to the level of vaccination in the wider population: individuals in the UK were risk compensating when rates of vaccination were rising. This effect was observed across four nations of the UK, each of which varied policies autonomously.

Can decision field theory enhance our understanding of health-based choices? Evidence from risky health behaviors.

Discrete choice models are almost exclusively estimated assuming random utility maximization (RUM) is the decision rule applied by individuals. Recent studies indicate alternative behavioral assumptions may be more appropriate in health. Decision field theory (DFT) is a psychological theory of decision-making, which has shown promise in transport research. This study introduces DFT to health economics, empirically comparing it to RUM and random regret minimization (RRM) in risky health settings, namely tobacco and vaccine choices. Model fit, parameter ratios, choice shares, and elasticities are compared between RUM, RRM and DFT. Test statistics for model differences are derived using bootstrap methods. Decision rule heterogeneity is investigated using latent class models, including novel latent class DFT models. Tobacco and vaccine choice data are better explained with DFT than with RUM or RRM. Parameter ratios, choice shares and elasticities differ significantly between models. Mixed results are found for the presence of decision rule heterogeneity. We conclude that DFT shows promise as a behavioral assumption that underpins the estimation of discrete choice models in health economics. The significant differences demonstrate that care should be taken when choosing a decision rule, but further evidence is needed for generalizability beyond risky health choices.

Harm reduction for smokers with little to no quit interest: can tobacco policies encourage switching to e-cigarettes?

OBJECTIVE: A pressing tobacco policy concern is how to help smokers who have little interest in quitting cigarettes, a group that often suffers severe health consequences. By switching from cigarettes to e-cigarettes, they could obtain nicotine, potentially with less harm. We examined if policy-relevant attributes of cigarettes/e-cigarettes might encourage these smokers to switch to e-cigarettes. METHODS: An online survey and discrete choice experiment on a nationally-representative sample of adult smokers in the US who reported low interest in quitting (n=2000). We modelled preference heterogeneity using a latent class, latent variable model. We simulated policies that could encourage switching to e-cigarettes. RESULTS: Participants formed two latent classes: (1) those with very strong preferences for their own cigarettes; and (2) those whose choices were more responsive to policies. The latter group's choices were only somewhat responsive to menthol cigarette bans and taxes; the former group's choices were unresponsive. CONCLUSIONS: The policies studied seem unlikely to encourage harm reduction for individuals with little interest in quitting smoking.

Malaysian public preferences and decision making for COVID-19 vaccination: A discrete choice experiment.

Background: Low vaccine uptake has the potential to seriously undermine COVID-19 vaccination programs, as very high coverage levels are likely to be needed for virus suppression to return life to normal. We aimed to determine the influence of vaccine attributes (including access costs) on COVID-19 vaccination preferences among the Malaysian public to improve national uptake. Methods: An online Discrete Choice Experiment (DCE) was conducted on a representative sample of 2028 Malaysians. Respondents were asked to make vaccination decisions in a series of hypothetical scenarios. A nested, mixed logit model was used to estimate the preferences for vaccination over vaccine refusal and for how those preferences varied between different sub-populations. The attributes were the risk of developing severe side effects of the vaccine, vaccine effectiveness, vaccine content, vaccination schedule, and distance from home to vaccination centre. Findings: Reported public uptake of COVID-19 vaccination was primarily influenced by the risk of developing severe side effects (b = -1·747, 95% CI = -2·269, -1·225), vaccine effectiveness (b = 3·061, 95% CI = 2·628, 3·494) and its Halal status (b = 3·722, 95% CI = 3·152, 4·292). Other factors such as appointment timing and travel distance to the vaccination centre also had an effect on vaccine uptake. There was substantial heterogeneity in preferences between different populations, particularly for age groups, ethnicity, regions, and underlying health conditions. Interpretation: Perceived effectiveness and side effects are likely to affect COVID-19 vaccine uptake in Malaysia. Halal content is critical to Malays' vaccination choices. Reducing the physical distance to vaccination centres, particularly in rural areas where uptake is lower, is likely to improve uptake. Funding: Ministry of Health Research Grant from the Malaysian government [NIH/800-3/2/1 Jld.7(46), grant reference no: 57377 and warrant no: 91000776].

Using stated-preferences methods to develop a summary metric to determine successful treatment of children with a surgical condition: a study protocol.

INTRODUCTION: Wide variation in the management of key paediatric surgical conditions in the UK has likely resulted in outcomes for some children being worse than they could be. Consequently, it is important to reduce unwarranted variation. However, major barriers to this are the inability to detect differences between observed and expected hospital outcomes based on the casemix of the children they have treated, and the inability to detect variation in significant outcomes between hospitals. A stated-preference study has been designed to estimate the value key stakeholders place on different elements of the outcomes for a child with a surgical condition. This study proposes to develop a summary metric to determine what represents successful treatment of children with surgical conditions. METHODS AND ANALYSIS: Preferences from parents, individuals treated for surgical conditions as infants/children, healthcare professionals and members of the public will be elicited using paired comparisons and kaizen tasks. A descriptive framework consisting of seven attributes representing types of operations, infections treated in hospital, quality of life and survival was identified. An experimental design has been completed using a D-efficient design with overlap in three attributes and excluding implausible combinations. All participants will be presented with an additional choice task including a palliative scenario that will be used as an anchor. The survey will be administered online. Primary analysis will estimate a mixed multinomial logit model. A traffic light system to determine what combination of attributes and levels represent successful treatment will be created. ETHICS AND DISSEMINATION: Ethics approval to conduct this study has been obtained from the Medical Sciences Inter-Divisional Research Ethics Committee (IDREC) at the University of Oxford (R59631/RE001-05). We will disseminate all of our results in peer-review publications and scientific presentations. Findings will be additionally disseminated through relevant charities and support groups and professional organisations.

Utility maximization versus regret minimization in health choice behavior: Evidence from four datasets.

Choice models in health are almost exclusively based on the neoclassical economic paradigm of utility maximization. Recently developed choice models have captured and shown empirical support for regret minimization as an alternative decision rule. In health economics, recent applications of RRM models indicate that individuals making health-based choices may exhibit regret minimization-type behavior. In this paper, we build on this research using a more flexible model that allows for heterogeneous decision rules, separately from preference heterogeneity, and comparing it to models that assume single decision rules. We use four datasets from diverse settings in which individuals make health choices: tobacco markets, genomic testing, and HIV prevention. We found that, if a one-size-fits-all rule is applied, then utility maximization was preferable to regret minimization for these datasets. However, we also find that individuals apply varying decision rules in similar proportions in these health settings, suggesting that models for heterogeneous decision rules were needed to capture these behaviors in these settings.

The Impact of Environmental Sustainability Labels on Willingness-to-Pay for Foods: A Systematic Review and Meta-Analysis of Discrete Choice Experiments.

Food production is a major contributor to environmental damage. More environmentally sustainable foods could incur higher costs for consumers. In this review, we explore whether consumers are willing to pay (WTP) more for foods with environmental sustainability labels ('ecolabels'). Six electronic databases were searched for experiments on consumers' willingness to pay for ecolabelled food. Monetary values were converted to Purchasing Power Parity dollars and adjusted for country-specific inflation. Studies were meta-analysed and effect sizes with confidence intervals were calculated for the whole sample and for pre-specified subgroups defined as meat-dairy, seafood, and fruits-vegetables-nuts. Meta-regressions tested the role of label attributes and demographic characteristics on participants' WTP. Forty-three discrete choice experiments (DCEs) with 41,777 participants were eligible for inclusion. Thirty-five DCEs (n = 35,725) had usable data for the meta-analysis. Participants were willing to pay a premium of 3.79 PPP$/kg (95%CI 2.7, 4.89, p ≤ 0.001) for ecolabelled foods. WTP was higher for organic labels compared to other labels. Women and people with lower levels of education expressed higher WTP. Ecolabels may increase consumers' willingness to pay more for environmentally sustainable products and could be part of a strategy to encourage a transition to more sustainable diets.

Weight loss interventions on health-related quality of life in those with moderate to severe obesity: Findings from an individual patient data meta-analysis of randomized trials.

The relationship between BMI and health-related quality of life (HRQoL) critically affects regulatory approval of interventions for weight loss, but evidence of the association is inconsistent. A higher standard of evidence than that available was sought with an IPD meta-analysis of 10,884 people enrolled in five randomized controlled trials of intentional weight loss interventions. Cross-sectional and longitudinal associations of BMI and HRQoL were estimated in mixed effects models specifying a latent variable for HRQoL. Spline regressions captured nonlinear associations across the range of BMI. In cross-sectional spline regressions, BMI was not associated with HRQoL for people with a BMI < 30 kg/m2 but was for those with a higher BMI. In longitudinal spline regressions, decreases in BMI were positively associated with HRQoL for people with a BMI ≥ 25 kg/m2 . The impact of change in BMI was larger for people with higher BMIs than for those with BMIs under 30 kg/m2 . Lower BMI and decreases in BMI were related to higher HRQoL for people living with obesity but not in the population without excess weight. HRQoL gains from weight loss are greater for more severe obesity. Commissioners should use these estimates for future decision making.

Toward the diagnosis of rare childhood genetic diseases: what do parents value most?

Genomic testing is becoming routine for diagnosing rare childhood genetic disease. Evidence underlying sustainable implementation is limited, focusing on short-term endpoints such as diagnostic yield, unable to fully characterize patient and family valued outcomes. Although genomic testing is becoming widely available, evidentiary and outcomes uncertainty persist as key challenges for implementation. We examine whether the current evidence base reflects public tolerance for uncertainty for genomics to diagnose rare childhood genetic disease. We conducted focus groups with general population parents in Vancouver, Canada, and Oxford, United Kingdom, to discuss expectations and concerns related to genomic testing to diagnose rare childhood genetic disease. Applying a purposive sampling technique, recruitment continued until thematic saturation was reached. Transcripts were analysed using thematic analysis. Thirty-three parents participated across four focus groups. Participants valued causal diagnoses alongside management strategies to improve patient health and wellbeing. Further, participants valued expanding the evidence base to reduce evidentiary uncertainty while ensuring security of information. Willingness to pay out of pocket for testing reflected perceived familial health benefit. Diagnostic yield fails to fully capture valued outcomes, and efforts to resolve uncertainty better reflect public priorities. Evaluations of genomic testing that fully integrate valued endpoints are necessary to ensure consistency with best practices and public willingness to accept the uncertain familial benefit.

Who, me? Optimism bias about US teenagers' ability to quit vaping.

BACKGROUND AND AIMS: The vaping rate among US teenagers has doubled in the last 2 years, which may be explained in part by teenagers' optimism that they would have relatively little trouble in quitting. The aim of this study was to estimate the extent to which teenagers exhibited optimism bias, what characteristics are associated with optimism bias and which factors are related to respondents' perceptions of how hard it would be for them to quit. DESIGN: A national, on-line, cross-sectional survey in 2018 using quota sampling. SETTING: United States. PARTICIPANTS: Respondents were 1610 teenagers aged 14-18 years who had ever tried or heard of JUULs or e-cigarettes generally. MEASUREMENTS: Optimism bias was defined as respondents' perceptions of their own difficulty quitting vaping compared with that of an average US person of their own age. Linear regression was used to examine associations between respondents' characteristics with both optimism bias and their own perceived difficulty quitting vaping. FINDINGS: More than 60% of teenagers were optimistically biased about their ability to quit vaping. Smoking (b = -0.69, P < 0.01) and JUULing (b = -0.62, P < 0.01) were negatively associated with optimism bias but reduced-price school lunch eligibility (0.27, P = 0.02) and school satisfaction were positively associated (b = 0.05, P = 0.02). Smoking (b = 0.85, P < 0.01) was associated with an increased perception of the difficulty of quitting. That association was negative for black respondents (b = -0.81, P = 0.01) and those eligible for reduced-priced lunches (b = -0.48, P = 0.01), and positive for Hispanic respondents (b = 0.47, P = 0.04). CONCLUSIONS: On average, US teenagers appear to show optimism bias about their ability to quit vaping, which decreases with smoking and vaping and increases with eligibility for reduced-price school lunches.