Should We Consider Including a Value for "Hope" as an Additional Benefit Within Health Technology Assessment?

Health technology assessment (HTA) typically uses average health-related quality of life gain as its main measure of benefit used in economic evaluation. Nevertheless, there have been calls to consider novel aspects of benefit including the "value of hope," defined as a patients' potential preferences for a wider distribution of treatment benefit with a positive skew, in the hope that they will be one of the lucky ones. The value of hope may also derive from feeling hopeful as a positive mental state, which may be missing from current measures of health-related quality of life. The value attributed to feeling hopeful could be related to, or additional to, the value derived from possible risk-seeking preferences. Here, we reflect upon the strength of the case for the inclusion of the "value of hope" taking a critical look at the commonly referenced evidence for including the "value of hope" as risk-seeking preferences. We also draw attention to other conceptions of hope-as an emotion, a cognitive process, or a combination of both-and reflect upon the potential of including these broader notions of hope into HTA. The case for the inclusion of the "value of hope" based on risk-seeking preferences is weak. We suggest research questions that could give further evidence on whether hope is an important missing value from HTA.

The EQ-HWB: Overview of the Development of a Measure of Health and Wellbeing and Key Results.

OBJECTIVES: Existing measures for estimating quality-adjusted life-years are mostly limited to health-related quality of life. This article presents an overview of the development the EQ-HWB (EQ Health and Wellbeing), which is a measure that encompasses health and wellbeing. METHODS: Stages: (1) Establishing domains through reviews of the qualitative literature informed by a conceptual framework. (2) Generation and selection of items to cover the domains. (3) Face validation of these items through qualitative interviews with 168 patients, social care users, general population, and carers across 6 countries (Argentina, Australia, China, Germany, United Kingdom, United States). (4) Extensive psychometric testing of candidate items (using classical, factor analysis, and item response theory methods) on > 4000 respondents in the 6 countries. Stakeholders were consulted throughout. RESULTS: A total of 32 subdomains grouped into 7 high-level domains were identified from the qualitative literature and 97 items generated to cover them. Face validation eliminated 36 items, modified 14, and added 3. Psychometric testing of 64 items found little difference in missing data or problems with response distribution, the conceptual model was confirmed except in China, and most items performed well in the item response theory in all countries. Evidence was presented to stakeholders in 2 rounds of consultation to inform the final selection of items for the EQ-HWB (25-item) and the short version of EQ-HWB (9-items). CONCLUSIONS: EQ-HWB measures have been developed internationally for evaluating interventions in health, public health, and social care including the impact on patients, social care users, and carers.

Valuing child and adolescent health: a qualitative study on different perspectives and priorities taken by the adult general public.

BACKGROUND: Quantitative health preference research has shown that different "perspectives", defined here as who is imagined to be experiencing particular health states, impact stated preferences. This qualitative project aimed to elucidate this phenomenon, within the context of adults' valuation of child and adolescent health states. METHODS: Six focus groups with 30 members of the UK adult public were conducted between December 2019 and February 2020 and analysed using framework analysis. Each focus group had two stages. First, participants individually completed time trade-off tasks and a pairwise task (mirroring a discrete choice experiment without duration) for two EQ-5D-Y health states, assuming a series of perspectives: (a) themselves at current age; (b) another adult; (c) 10-year old child; (d) themselves as a 10-year old child. Second, a semi-structured discussion explored their responses. RESULTS: Participants' views were often heterogeneous, with some common themes. Qualitatively, participants expressed a different willingness to trade-off life years for a 10-year old child versus themselves or another adult, and this differed by the health profile and child imagined. The same health states were often viewed as having a different impact on utility for a 10-year old child than adults. Imagining a 10-year old child is difficult and there is variation in who is imagined. Participants found answering based on their own-adult perspective most acceptable. There were no strong preferences for prioritising child health over working-age adults' health. CONCLUSIONS: If an adult sample is used to value child- and adolescent-specific health states it is important to consider the perspective employed. Members of the adult public provide different responses when different perspectives are used due to differences in the perceived impact of the same health states. If adults are asked to imagine a child, we recommend that sampling is representative for parental status, since this can affect preferences.

Comparing aversions to outcome inequality and social risk in health and income: An empirical analysis using hypothetical scenarios with losses.

Evaluation of future social welfare may not only depend on the aggregate of individual prospects, but also on how the prospects are distributed across individuals. The latter in turn would depend on how people perceive inequality and risk at the collective level (or "social risk"). This paper examines distributional preferences regarding inequality in outcomes and social risk for health and income in the context of losses. Specifically, four kinds of aversions are compared, (a) outcome-inequality aversion in health, (b) outcome-inequality aversion in income, (c) social-risk aversion in health, (d) and social-risk aversion in income. Face-to-face interviews of a representative general public sample in Spain are undertaken using hypothetical scenarios involving losses in health or income across otherwise equal groups. Aversion parameters are compared assuming social welfare functions with constant relative or constant absolute aversion. We find that in both domains, outcome-inequality aversion and social-risk aversion are not the same; and that neither aversion is the same across the two domains. Outcome-inequality aversion in income is the strongest, followed by social-risk aversion in income and social-risk aversion in health, and outcome-inequality aversion in health coming last, where most of these are statistically significantly different from each other.

E-learning and health inequality aversion: A questionnaire experiment.

In principle, questionnaire data on public views about hypothetical trade-offs between improving total health and reducing health inequality can provide useful normative health inequality aversion parameter benchmarks for policymakers faced with real trade-offs of this kind. However, trade-off questions can be hard to understand, and one standard type of question finds that a high proportion of respondents-sometimes a majority-appear to give exclusive priority to reducing health inequality. We developed and tested two e-learning interventions designed to help respondents understand this question more completely. The interventions were a video animation, exposing respondents to rival points of view, and a spreadsheet-based questionnaire that provided feedback on implied trade-offs. We found large effects of both interventions in reducing the proportion of respondents giving exclusive priority to reducing health inequality, though the median responses still implied a high degree of health inequality aversion and-unlike the video-the spreadsheet-based intervention introduced a substantial new minority of non-egalitarian responses. E-learning may introduce as well as avoid biases but merits further research and may be useful in other questionnaire studies involving trade-offs between conflicting values.

An exploration of the non-iterative time trade-off method to value health states.

Time Trade-Off (TTO) usually relies on "iteration," which is susceptible to bias. Discrete Choice Experiment with duration (or DCETTO ) is free of such bias, but respondents find this cognitively more challenging. This paper explores non-iterative TTO with or without lead time: NI(LT)TTO. In NI(LT)TTO, respondents see a series of independent pairwise choices without iteration (similar to DCETTO ), but one of the two scenarios always involves full health for a shorter duration (similar to TTO). We compare three different "types" of NI(LT)TTO relative to DCETTO . Each type is presented in two "modes": (a) verbally tabulated (as in a DCE) and (b) with visual aids (as in a TTO). The study has 8 survey variants, each with 12 experimental choice tasks and a 13th task with a logically determined answer. Data on the 12 experimental choices from an online survey of 6,618 respondents are modelled, by variant, using conditional logistic regressions. The results indicate that NI(LT)TTO is feasible, but some relatively mild states appear to have implausibly low predicted values, and the range of predicted values is much narrower than in DCETTO . The presentation of NI(LT)TTO tasks needs further improvement.

Re-Thinking 'The Different Perspectives That can be Used When Eliciting Preferences in Health'.

The 2003 Health Economics paper by Dolan, Olsen, Menzel and Richardson on 'An inquiry into the different perspectives that can be used when eliciting preferences in health' presents a conceptual framework of six perspectives along two dimensions: preferences (personal, social, and socially inclusive personal) and context (ex ante and ex post). The objective of our paper is to re-think this framework. We ask four questions concerning: the patient, or the user of the treatment; the payer of the treatment; the assessor of the value of treatment; and the timing of the illness and the nature of its risk. These questions refine the preference and context dimensions, and lead to the identification of perspectives not classified by the original framework. We propose an extended framework with five preferences (personal, non-use, proxy, social, and socially inclusive personal) and five contexts (one of which is ex post and four ex ante): since two of these cells are empty, this results in 23 possible perspectives. Online Supplementary Information presents 11 of these more formally to clearly distinguish between them and uses monetary and non-monetary (time trade-off) valuation tasks as examples. Copyright © 2017 John Wiley & Sons, Ltd.

Eliciting the Level of Health Inequality Aversion in England.

Health inequality aversion parameters can be used to represent alternative value judgements about policy concern for reducing health inequality versus improving total health. In this study, we use data from an online survey of the general public in England (n = 244) to elicit health inequality aversion parameters for both Atkinson and Kolm social welfare functions. We find median inequality aversion parameters of 10.95 for Atkinson and 0.15 for Kolm. These values suggest substantial concern for health inequality among the English general public which, at current levels of quality adjusted life expectancy, implies weighting health gains to the poorest fifth of people in society six to seven times as highly as health gains to the richest fifth. Copyright © 2016 John Wiley & Sons, Ltd.

An exploratory study to test the impact on three "bolt-on" items to the EQ-5D.

BACKGROUND: Generic preference-based measures were criticized for being inappropriate in some conditions. One solution is to include "bolt-on" dimensions describing additional specific health problems. OBJECTIVES: This study aimed to develop bolt-on dimensions to the EuroQol five-dimensional questionnaire (EQ-5D) and assess their impact on health state values. METHODS: Bolt-on dimensions were developed for vision problems, hearing problems, and tiredness. Each bolt-on dimension had three severity levels to match the EQ-5D. Three "core" EQ-5D states across a range of severity were selected, and each level of a bolt-on item was added, resulting in nine states in each condition. Health states with and without the bolt-on dimensions were valued by 300 members of the UK general public using time trade-off in face-to-face interviews, and mean health state values were compared using t tests. Regression analysis examined the impact of the bolt-on variants and the level of the bolt-on items after controlling for sociodemographic characteristics. RESULTS: Bolt-on dimensions had an impact on health state values of the EQ-5D; however, the size, direction, and significance of the impact depend on the severity of the core EQ-5D state and of the bolt-on dimension. Regression analysis demonstrated that after controlling for possible differences in sociodemographic characteristics between the groups, there were no significant differences in health state values between the three bolt-on dimensions but confirmed that the impact depended on the severity of the EQ-5D health state and the levels of bolt-on dimensions. CONCLUSIONS: The impact of a bolt-on dimension on the EQ-5D depends on the core health state and the level of the bolt-on dimension. Further research in this area is encouraged.

Measuring quality of life in children with speech and language difficulties: a systematic review of existing approaches.

BACKGROUND: Children's and adolescent's speech and language difficulties (SaLD) can affect various domains of quality of life (QoL), and speech and language therapy interventions are critical to improving QoL. Systematically measuring QoL outcomes in this population is highly complex due to factors such as heterogeneity in impairments and differing targets during intervention. However, measurements of QoL are increasingly required by healthcare commissioners and policy-makers to inform resource allocation. AIMS: To review the use of QoL measures in research involving children (age ≤ 18 years) with SaLD. METHODS & PROCEDURES: A systematic review was undertaken. A systematic search across various databases was performed. Information on the methodological details of each relevant study, along with descriptions of the QoL measures employed, were extracted into standardized data extraction forms. Findings were discussed in a narrative synthesis. OUTCOMES & RESULTS: Twenty-one relevant studies were identified that deal with a range of subpopulations of children with SaLD. For the most part, generic QoL measures were used, although there was little convergence on the type of QoL measures employed throughout the literature. Five studies utilized preference-based QoL measures, including the 16D/17D, HUI3, EQ-5D and QWB-SA. Of these measures, the HUI3 demonstrated the most promising discriminant validity, although the preference weights for this measure were generated with adults. CONCLUSIONS & IMPLICATIONS: QoL among children with SaLD is not yet being captured in a systematic way. The HUI3 measure appears to show some promise for generating relevant preference-based QoL estimates, although further testing of the measure is required.

Egalitarianism and altruism in health: some evidence of their relationship.

BACKGROUND: Egalitarianism and altruism are two ways in which people may have attitudes that go beyond the narrowly defined selfish preferences. The theoretical constructs of egalitarianism and altruism are different from each other, yet there may be connections between the two. This paper explores the empirical relationship between egalitarianism and altruism, in the context of health. METHODS: We define altruism as individual behaviour that aims to benefit another individual in need; and egalitarianism as a characteristic of a social welfare function, or a meta-level preference. Furthermore, we specify a model that explains the propensity of an individual to be egalitarian in terms of altruism and other background characteristics. Individuals who prefer a hypothetical policy that reduces socioeconomic inequalities in health outcomes over another that does not are regarded 'egalitarian' in the health domain. On the other hand, 'altruism' in the health context is captured by whether or not the same respondents are (or have been) regular blood donors, provided they are medically able to donate. Probit models are specified to estimate the relationship between egalitarianism and altruism, thus defined. A representative sample of the Spanish population was interviewed for the purpose (n = 417 valid cases). RESULTS: Overall, 75% of respondents are found to be egalitarians, whilst 35% are found to be altruists. We find that, once controlled for background characteristics, there is a statistically significant empirical relationship between egalitarianism and altruism in the health context. On average, the probability of an altruist individual supporting egalitarianism is 10% higher than for a non-altruist person. Regarding the other control variables, those living in high per capita income regions have a lower propensity and those who are politically left wing have a higher propensity to be an egalitarian. We do not find evidence of a relationship between egalitarianism and age, socioeconomic status or religious practices. CONCLUSION: Altruist individuals have a higher probability to be egalitarians than would be expected from their observed background characteristics.

Binary choice health state valuation and mode of administration: head-to-head comparison of online and CAPI.

BACKGROUND: Health state valuation exercises can be conducted online, but the quality of data generated is unclear. OBJECTIVE: To investigate whether responses to binary choice health state valuation questions differ by administration mode: online versus face to face. METHODS: Identical surveys including demographic, self-reported health status, and seven types of binary choice valuation questions were administered in online and computer-assisted personal interview (CAPI) settings. Samples were recruited following procedures employed in typical online or CAPI studies. Analysis included descriptive comparisons of the distribution of responses across the binary options and probit regression to explain the propensity to choose one option across modes of administration, controlling for background characteristics. RESULTS: Overall, 422 (221 online; 201 CAPI) respondents completed a survey. There were no overall age or sex differences. Online respondents were educated to a higher level than were the CAPI sample and general population, and employment status differed. CAPI respondents reported significantly better general health and health/life satisfaction. CAPI took significantly longer to complete. There was no effect of the mode of administration on responses to the valuation questions, and this was replicated when demographic differences were controlled. CONCLUSIONS: The findings suggest that both modes may be equally valid for health state valuation studies using binary choice methods (e.g., discrete choice experiments). There are some differences between the observable characteristics of the samples, and the groups may differ further in terms of unobservable characteristics. When designing health state valuation studies, the advantages and disadvantages of both approaches must be considered.

A comparison of alternative variants of the lead and lag time TTO.

'Lead Time' TTO improves upon conventional TTO by providing a uniform method for eliciting positive and negative values. This research investigates (i) the values generated from different combinations of time in poor health and in full health; and the order in which these appear (lead vs. lag); (ii) whether values concur with participants' views about states; (iii) methods for handling extreme preferences. n = 208 participants valued five EQ-5D states, using two of four variants. Combinations of lead time and health state duration were: 10 years and 20 years; 5 years and 1 year; 5 years and 10 years; and a health state duration of 5 years with a lag time of 10 years. Longer lead times capture more preferences, but may involve a framing effect. Lag time results in less non-trading for mild states, and less time being traded for severe states. Negative values broadly agree with participants' stated opinion that the state is worse than dead. The values are sensitive to the ratio of lead time to duration of poor health, and the order in which these appear (lead vs. lag). It is feasible to handle extreme preferences though challenges remain.

The cost of hypertension-related ill-health attributable to environmental noise.

Hypertension (HT) is associated with environmental noise exposure and is a risk factor for a range of health outcomes. The study aims were to identify key HT related health outcomes and to quantify and monetize the impact on health outcomes attributable to environmental noise-related HT. A reiterative literature review identified key HT related health outcomes and their quantitative links with HT. The health impact of increases in environmental noise above recommended daytime noise levels (55 dB[A]) were quantified in terms of quality adjusted life years and then monetized. A case study evaluated the cost of environmental noise, using published data on health risks and the number of people exposed to various bands of environmental noise levels in the United Kingdom (UK). Three health outcomes were selected based on the strength of evidence linking them with HT and their current impact on society: Acute myocardial infarction (AMI), stroke and dementia. In the UK population, an additional 542 cases of HT-related AMI, 788 cases of stroke and 1169 cases of dementia were expected per year due to daytime noise levels ≥55 dB(A). The cost of these additional cases was valued at around £1.09 billion, with dementia accounting for 44%. The methodology is dependent on the availability and quality of published data and the resulting valuations reflect these limitations. The estimated intangible cost provides an insight into the scale of the health impacts and conversely the benefits that the implementation of policies to manage environmental noise may confer.

Valuing states from multiple measures on the same visual analogue sale: a feasibility study.

Previous methods of empirical mapping involve using regressions on patient or general population self-reported data from datasets involving two or more measures. This approach relies on overlap in the descriptive systems of the measures and assumes it is appropriate to use different measures on the same population, which may not always be the case. This paper presents a feasibility study for a new approach to mapping between preference-based measures (PBM) using general population visual analogue scale (VAS) values as a common yardstick. We use data from a valuation study of 502 members of the UK general population, where, using ranking and VAS tasks, interviewees simultaneously valued health states defined by three of six PBM: EQ-5D (generic), SF-6D (generic), HUI2 (generic for children and adults), AQL-5D (asthma specific), OPUS (social care specific) and ICECAP (capabilities). Regression techniques are used to estimate the relationship between these VAS values and the original value set (i.e. 'tariff'). These results are subsequently used to estimate the relationship between all six PBM to enable 'value-based mapping' between measures. This new method of mapping potentially has a useful role in evidence synthesis and cross programme comparisons in studies using different measures.

Public preferences for responsibility versus public preferences for reducing inequalities.

In cost-utility analysis, the numbers of quality-adjusted life years (QALYs) gained are aggregated by placing the same weight on each QALY. Deviations from this rubric have been proposed on a number of grounds, including the degree to which persons might be deemed responsible for the illness faced, and inequality in lifetime health between groups. Most research has looked at these factors in isolation. This paper analyses public preferences about the relative importance of these factors. Over 500 members of the general public in the UK are interviewed in their homes. Where "blameworthy" groups experience a moderate drop in quality of life due to their behaviour, they appear to receive higher priority than an otherwise "trustworthy" group if they also experience poorer health prospects because the latter is weighted more heavily than the former.

Using mixed methods research to explore the effect of an adaptation exercise on general population valuations of health states.

PURPOSE: To understand the effect of an adaptation exercise (AE) on general population values for rheumatoid arthritis (RA) states. METHODS: A sequential mixed methods design was employed: an analysis of a dataset to develop RA states for valuing in later phases of the study; a qualitative interview study with members of the general population to identify how an AE affected valuing of the RA states and to help design a questionnaire for the final phase; and a quantitative quasi-experimental study to identify factors that influence change in values after being informed about adaptation. RESULTS: Three RA states were developed using Rasch and cluster analyses. Participants in the qualitative phase identified a range of ways in which information about adaptation affected their values. For example, they realized they could adapt to RA because their family and friends who had RA, or similar conditions, could cope. A 25-item questionnaire was developed and used during the final phase to identify that younger and healthier individuals were more likely to increase their values after being informed about disease adaptation. The qualitative findings were revisited and found to support the quantitative results. CONCLUSIONS: This approach facilitated understanding of whether and how an AE affected valuing of health states. Each phase affected the next phase of the study, leading to the conclusion that general population respondents who have little experience of disease will likely increase their health state values after being informed about adaptation because they understand that they could cope with the disease.

Do they just know more, or do they also have different preferences? An exploratory analysis of the effects of self-reporting serious health problems on health state valuation.

BACKGROUND: Health state valuation is often conducted by people valuing either only their own health state (experience-based valuation) or several stylised states (hypothetical valuation). The approach used can affect the elicited values, but it is not clear whether this is caused by different understandings of the states (an "information mechanism") and/or by different opinions of the states (a "preference mechanism"). Justifying privileging the valuations of those with the relevant health state experience solely because they are better informed is insufficient when their opinions differ. This study proposes a new framework to examine the effect of having health problems on health state valuation by distinguishing "within-dimensional" effects (which can be due to better information or due to differences in opinion) and "cross-dimensional" effects (which must be due to differences in opinion). METHODS: Secondary data from the UK that valued EQ-5D-5L using Discrete Choice Experiments with duration (DCETTO) are remodelled controlling for whether a respondent self-reports serious (viz., severe or extreme) problems in "pain or discomfort" (PD) or "anxiety or depression" (AD). The main analysis uses respondents who have serious PD or serious AD alongside matched respondents who do not, and assumes constant proportional time trade-off. RESULTS: Self-reporting serious PD or serious AD problems has no within-dimensional effect on health state preferences. However, self-reporting serious AD problems has negative cross-dimensional effects on the utility of having any problem in PD, which suggests that the preference mechanism is present. A similar pattern holds when all available (unmatched) data are used and when constant proportional time trade-off is relaxed. CONCLUSIONS: Findings consistent with the preference mechanism indicate that those with serious health problems may have different opinions on the value of health states compared to the rest of the population, which has normative implications for the debate on which values to use.

Behavioral Theories That Have Influenced the Way Health State Preferences Are Elicited and Interpreted: A Bibliometric Mapping Analysis of the Time Trade-Off Method With VOSviewer Visualization.

Aim: The aim of this paper is to develop an understanding of how behavioral theories have influenced the way preferences for health-related quality of life are elicited and interpreted. We focus on the Time Trade-off (TTO) method given it represents the quality-adjusted life-year (QALY) concept-that survival in less-than-full health can be deemed equivalent to a shorter survival in full health. To our knowledge this is the first review using a combination of systematic scoping review, bibliometrics and VOSviewer visualization to map the development of ideas in health economics. Methods: A priori, we selected three behavioral theories to explore within our review, referred to here as Expected Utility Theory, Non-Expected Utility Theory and Probabilistic Choice Theory. A fourth topic, Order Effects, is defined broadly to encompass behavioral theories around timing/sequence of events. For the main search, Scopus was used to identify literature that had (a) elicited TTO values and/or (b) contributed to the way TTO values were elicited and interpreted, from inception to July 2021. Papers that focused on the latter category were given the label "behavioral" and underwent additional analyses. A two stage-screening was applied to assess eligibility. Co-citation, co-authorship and co-occurrence of keywords was used to chart the development of TTO over time. Results: A total of 1,727 records were retrieved from Scopus and were supplemented by an additional 188 papers. There were 856 applied and 280 behavioral papers included in the final corpus, with the behavioral set split equally into four sets of 70 papers to chart the development of keywords over time: (1) 1972-1999; (2) 2000-2010, (3) 2010-2015 and (4) 2015-2021. Discussion: The keyword analysis suggested that whilst some ideas transition quickly from economic theory to the TTO literature, such as the impact of Order Effects, others take longer to be assimilated, for example Non-Expected Utility models or failure of constant discounting. It is therefore important that researchers within health economics work more closely with those in mainstream economics and keep abreast of the wider economics and behavioral sciences to expedite the uptake of new and relevant ideas.

Application of evoked response audiometry for specifying aberrant gamma oscillations in schizophrenia.

Gamma oscillations probed using auditory steady-state response (ASSR) are promising clinical biomarkers that may give rise to novel therapeutic interventions for schizophrenia. Optimizing clinical settings for these biomarker-driven interventions will require a quick and easy assessment system for gamma oscillations in psychiatry. ASSR has been used in clinical otolaryngology for evoked response audiometry (ERA) in order to judge hearing loss by focusing on the phase-locked response detectability via an automated analysis system. Herein, a standard ERA system with 40- and 46-Hz ASSRs was applied to evaluate the brain pathophysiology of patients with schizophrenia. Both ASSRs in the ERA system showed excellent detectability regarding the phase-locked response in healthy subjects and sharply captured the deficits of the phase-locked response caused by aberrant gamma oscillations in individuals with schizophrenia. These findings demonstrate the capability of the ERA system to specify patients who have aberrant gamma oscillations. The ERA system may have a potential to serve as a real-world clinical medium for upcoming biomarker-driven therapeutics in psychiatry.