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 QALY at 50: One story many voices.

Research on quality adjusted life year (QALY) has been underway for just over 50 years, which seems like a suitable milestone to review its history. The purpose of this study is to provide a historical overview of why the QALY was developed, the key theoretical work undertaken by Torrance, Bush and Fanshel and how two seminal papers shaped its subsequent development. Moving the QALY forward - there are several historical and reflective exercises. The historical interplay between politics, policy and the challenges facing the National Health Service (NHS) in formulating the QALY concept in the UK has been explored in some depth already, whilst the conceptualization and development of the methodological framework is relatively underexplored. We address this gap by viewing the QALY through the lens of the methodological debates, reflecting upon two key papers underpinning the QALY methodology and how these methods have been developed over time. In part the changes in technology e.g. Google Scholar, and the availability of tools to search for early uses of the QALY allow us to better understand the historical context in which the theoretical development of the QALY has taken place. Here we celebrate two seminal papers that shaped early QALY development. The first section provides a history of these papers, summaries their contributions and explores the uptake of these papers over time. The second section reviews the methodological debates that have surrounded the QALY over the last 50 years and looks at how the QALY has moved to address these challenges. The third section presents the voices of diverse commentators representing the field of health economics who have contributed to the subsequent development of the QALY in both theoretical and empirical capacities and captures their thoughts about future research and policy use of QALYS.

Equity in healthcare access and service coverage for older people: a scoping review of the conceptual literature.

There is currently no global review of the conceptual literature on the equity of healthcare coverage (including access) for older people. It is important to understand the factors affecting access to health and social care for this group, so that policy and service actions can be taken to reduce potential inequities. A scoping review of published and grey literature was conducted with the aim of summarising how health and social care service access and coverage for older people has been conceptualised. PubMed, MEDLINE, PsycINFO, CINAHL, Web of Science, SciELO, LILACS, BIREME and Global Index Medicus were searched. Selection of sources and data charting were conducted independently by two reviewers. The database searches retrieved 10 517 citations; 32 relevant articles were identified for inclusion from a global evidence base. Data were summarised and a meta-framework and model produced listing concepts specific to equitable health and social care service coverage relating to older people. The meta-framework identified the following relevant factors: acceptability, affordability, appropriateness, availability and resources, awareness, capacity for decision-making, need, personal social and cultural circumstances, physical accessibility. This scoping review is relevant to the development and specification of policy for older people. It conceptualises those factors, such as acceptability and affordability, that affect an older person's ability and capacity to access integrated, person-centred health and social care services in a meaningful way. These factors should be taken into account when seeking to determine whether equity in service use or access is being achieved for older people.

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.

What is the best approach to adopt for identifying the domains for a new measure of health, social care and carer-related quality of life to measure quality-adjusted life years? Application to the development of the EQ-HWB?

Economic evaluation combines costs and benefits to support decision-making when assessing new interventions using preference-based measures to measure and value benefits in health or health-related quality of life. These health-focused instruments have limited ability to capture wider impacts on informal carers or outcomes in other sectors such as social care. Sector-specific instruments can be used but this is problematic when the impact of an intervention straddles different sectors.An alternative approach is to develop a generic preference-based measure that is sufficiently broad to capture important cross-sector outcomes. We consider the options for the selection of domains for a cross-sector generic measure including how to identify domains, who should provide information on the domains and how this should be framed. Beyond domain identification, considerations of criteria and stakeholder needs are also identified.This paper sets out the case for an approach that relies on the voice of patients, social care users and informal carers as the main source of domains and describes how the approach was operationalised in the 'Extending the QALY' project which developed the new measure, the EQ-HWB (EQ health and wellbeing instrument). We conclude by discussing the strengths and limitations of this approach. The new measure should be sufficiently generic to be used to consistently evaluate health and social care interventions, yet also sensitive enough to pick up important changes in quality of life in patients, social care users and carers.

Does the UK-public's aversion to inequalities in health differ by group-labelling and health-gain type? A choice-experiment.

Public health policy has two primary aims: promoting population health and reducing health inequalities. When these aims conflict, policy-makers must determine the relative importance to place on each in decision-making. We conducted a computer-based, face-to-face, choice-experiment to explore how the UK-public think government should act in these situations; and to explore how "inequality-aversion" may differ depending on the groups between which a health inequality exists and type of health an intervention provides. We tested three hypotheses: (1) the UK-public are more averse to inequalities in health between socioeconomic groups than they are to inequalities in health between neutrally labelled groups; (2) this difference is, at least in part, driven by the role non-health information plays in determining aversion to inequalities in health between socioeconomic groups; and (3) the UK-public are more willing to prioritise groups with lower lifetime health over groups with higher lifetime health if an intervention improves life-expectancy than if it improves quality-of-life. Eighty people participated in Sheffield and Hull in May/June 2019. Each participant completed three Person-Trade-Off exercises between interventions that would improve population health and reduce health inequalities, or improve population health by a larger amount but increase health inequalities. Participants were randomised to exercises involving scenarios with socioeconomic groups or neutrally-labelled groups, and each answered questions about three health-benefit types: increased life-expectancy; pain-relief; and mobility-improvement. Following the exercises, participants provided rationales for their selections. Respondents were (1) more averse to inequalities in health between socioeconomic groups than neutrally labelled groups. Participant rationales suggest (2) this divergence is partly motivated by factors other than health: for example, financial inequality between socioeconomic groups. The sample was also (3) more willing to prioritise neutrally labelled groups with lower lifetime health if an intervention improves life-expectancy rather than if it improves quality-of-life.

Implausible States: Prevalence of EQ-5D-5L States in the General Population and Its Effect on Health State Valuation.

The EQ-5D is made up of health state dimensions and levels, in which some combinations seem less "plausible" than others. If "implausible" states are used in health state valuation exercises, then respondents may have difficulty imagining them, causing measurement error. There is currently no standard solution: some valuation studies exclude such states, whereas others leave them in. This study aims to address 2 gaps in the literature: 1) to propose an evidence-based set of the least prevalent two-way combinations of EQ-5D-5L dimension levels and 2) to quantify the impact of removing perceived implausible states from valuation designs. For the first aim, we use data from 2 waves of the English General Practitioner Patient Survey (n = 1,639,453). For the second aim, we remodel a secondary data set of a Discrete Choice Experiment (DCE) with duration that valued EQ-5D-5L and compare across models that drop observations involving different health states: 1) implausible states as defined in the literature, 2) the least prevalent states identified in stage 1, and 3) randomly select states, alongside 4) a model that does not drop any observations. The results indicate that two-way combinations previously thought to be implausible actually exist among the general population; there are other combinations that are rarer, and removing implausible states from an experimental design of a DCE with duration leads to value sets with potentially different characteristics depending on the criterion of implausible states. We advise against the routine removal of implausible states from health state valuation studies.

How averse are the UK general public to inequalities in health between socioeconomic groups? A systematic review.

There is growing interest in the use of "distributionally-sensitive" forms of economic evaluation that capture both the impact of an intervention upon average population health and the distribution of that health amongst the population. This review aims to inform the conduct of distributionally sensitive evaluations in the UK by answering three questions: (1) How averse are the UK public towards inequalities in lifetime health between socioeconomic groups? (2) Does this aversion differ depending upon the type of health under consideration? (3) Are the UK public as averse to inequalities in health between socioeconomic groups as they are to inequalities in health between neutrally framed groups? EMBASE, MEDLINE, EconLit, and SSCI were searched for stated preference studies relevant to these questions in October 2017. Of the 2155 potentially relevant papers identified, 15 met the predefined hierarchical eligibility criteria. Seven elicited aversion to inequalities in health between socioeconomic groups, and eight elicited aversion between neutrally labelled groups. We find general, although not universal, evidence for aversion to inequalities in lifetime health between socioeconomic groups, albeit with significant variation in the strength of that preference across studies. Second, limited evidence regarding the impact of the type of health upon aversion. Third, some evidence that the UK public are more averse to inequalities in lifetime health when those inequalities are presented in the context of socioeconomic inequality than when presented in isolation.

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.

The SIPHER Consortium: Introducing the new UK hub for systems science in public health and health economic research.

The conditions in which we are born, grow, live, work and age are key drivers of health and inequalities in life chances. To maximise health and wellbeing across the whole population, we need well-coordinated action across government sectors, in areas including economic, education, welfare, labour market and housing policy. Current research struggles to offer effective decision support on the cross-sector strategic alignment of policies, and to generate evidence that gives budget holders the confidence to change the way major investment decisions are made. This open letter introduces a new research initiative in this space. The SIPHER ( Systems Science in Public Health and Health Economics Research) Consortium brings together a multi-disciplinary group of scientists from across six universities, three government partners at local, regional and national level, and ten practice partner organisations. The Consortium's vision is a shift from health policy to healthy public policy, where the wellbeing impacts of policies are a core consideration across government sectors. Researchers and policy makers will jointly tackle fundamental questions about: a) the complex causal relationships between upstream policies and wellbeing, economic and equality outcomes; b) the multi-sectoral appraisal of costs and benefits of alternative investment options; c) public values and preferences for different outcomes, and how necessary trade-offs can be negotiated; and d) creating the conditions for intelligence-led adaptive policy design that maximises progress against economic, social and health goals. Whilst our methods will be adaptable across policy topics and jurisdictions, we will initially focus on four policy areas: Inclusive Economic Growth, Adverse Childhood Experiences, Mental Wellbeing and Housing.

Striving for a Societal Perspective: A Framework for Economic Evaluations When Costs and Effects Fall on Multiple Sectors and Decision Makers.

In most societies, resources are distributed by individuals acting in markets and by governments through some form of collective decision-making process. Economic evaluation offers a set of tools to inform collective decisions by examining the resource requirements and outcomes of alternative policies. The 'societal perspective' has been advocated, but less consideration has been given to what this should include and its practical implementation. This paper presents a framework for economic evaluation of policies with costs and outcomes falling on different sectors (e.g. health, criminal justice, education) and involving different decision makers. It extends the 'impact inventory' developed by the Second Panel on Cost-Effectiveness in Health and Medicine by considering all affected individuals and reflecting how outcomes attributed to an intervention can be compared with outcomes forgone as a result of resources not being available for other purposes. The framework sets out the series of assessments to be made, distinguishing points at which value judgements feed into the evaluation, and the implications of alternative judgements. These assessments reflect the institutional arrangements of public bodies, for example, their funding, the outcomes they consider important and their relative valuations of these outcomes. By avoiding the use of an abstract 'societal perspective', the contribution of the framework is to inform multiple decision makers with different objectives and provide practical guidance on overall societal impact.

Equal access for equal need: Eliciting public preferences for access to health treatment by employment status.

The National Health Service in the UK is set up under the principle of "equal access for equal need", where those with identical medical needs should be given equal priority in receiving health care. However, non-medical needs may also be relevant in health care decision-making. This paper considers how members of the general public value access to a health service given equal medical needs, where some service users have additional non-medical needs. There are three primary research questions. First, are public preferences regarding access to a health care service symmetric and inequality averse? Second, are public preferences asymmetric across different needs groups? And third, which individual characteristics of respondents are predictive of different public preferences in this domain? An online survey of the UK general public was conducted in January 2017 using binary choice questions. The hypothetical scenarios involved allocating extra resources from a social perspective, to reduce the waiting time to access a mental health service for the unemployed, for the employed, or for both groups. Based on a valid sample of 662 respondents, the study found that the three main preference categories were: inequality averse and symmetric, inequality averse and asymmetric in favour of the unemployed, and inequality seeking and asymmetric in favour of the unemployed, with the first group being the largest. Respondents' current labour market status was found to explain their preferences so that those who were currently job-seeking were more likely to demonstrate preferences that favoured the unemployed, and those who were currently unemployed were less likely to demonstrate asymmetric preferences that favoured the employed. The implications from these findings are that health policies in the UK that support equal access for equal medical need are likely to be received most favourably, yet a non-trivial minority may support policies favouring those with other, non-medical needs.

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.

Valuing health at the end of life: A review of stated preference studies in the social sciences literature.

A source of debate in the health care priority setting literature is whether to weight health gains to account for equity considerations, such as concern for those with very short life expectancy. This paper reviews the empirical evidence in the published social sciences literature relevant to the following research question: do members of the public wish to place greater weight on a unit of health gain for end-of-life patients than on that for other types of patients? An electronic search of the Social Sciences Citation Index for articles published until October 2017 was conducted, with follow-up of references to obtain additional data. Hierarchical criteria were applied to select empirical studies reporting stated preferences relating to hypothetical health care priority setting contexts. Twenty-three studies met the inclusion criteria and were included in the review. Choice exercises were the most common method used to elicit preferences; other approaches included budget allocation, person trade-off and willingness-to-pay. Some studies found that observed preferences regarding end-of-life patients are influenced by information about the patients' ages. Overall, the evidence is mixed, with eight studies that report evidence consistent with a 'premium' for end-of-life treatments and 11 studies that do not. Methodological and design aspects that appear to influence the findings of end-of-life-related preference studies are identified and discussed. The findings of the UK studies have particular relevance for assessing the legitimacy of the National Institute for Health and Care Excellence's policy for appraising life-extending end-of-life treatments.

How Robust Are Value Judgments of Health Inequality Aversion? Testing for Framing and Cognitive Effects.

BACKGROUND: Empirical studies have found that members of the public are inequality averse and value health gains for disadvantaged groups with poor health many times more highly than gains for better off groups. However, these studies typically use abstract scenarios that involve unrealistically large reductions in health inequality and face-to-face survey administration. It is not known how robust these findings are to more realistic scenarios or anonymous online survey administration. METHODS: This study aimed to test the robustness of questionnaire estimates of inequality aversion by comparing the following: 1) small versus unrealistically large health inequality reductions, 2) population-level versus individual-level descriptions of health inequality reductions, 3) concrete versus abstract intervention scenarios, and 4) online versus face-to-face mode of administration. Fifty-two members of the public participated in face-to-face discussion groups, while 83 members of the public completed an online survey. Participants were given a questionnaire instrument with different scenario descriptions for eliciting aversion to social inequality in health. RESULTS: The median respondent was inequality averse under all scenarios. Scenarios involving small rather than unrealistically large health gains made little difference in terms of inequality aversion, as did population-level rather than individual-level scenarios. However, the proportion expressing extreme inequality aversion fell 19 percentage points when considering a specific health intervention scenario rather than an abstract scenario and was 11 to 21 percentage points lower among online public respondents compared with the discussion group. CONCLUSIONS: Our study suggests that both concrete scenarios and online administration reduce the proportion expressing extreme inequality aversion but still yield median responses that imply substantial health inequality aversion.

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.

Exploring a new method for deriving the monetary value of a QALY.

Several studies have sought to determine the monetary value of health gains expressed as quality adjusted life years (QALYs) gained, predominantly using willingness to pay approaches. However, willingness to pay has a number of recognized problems, most notably its insensitivity to scope. This paper presents an alternative approach to estimate the monetary value of a QALY, which is based on the time trade-off method. Moreover, it presents the results of an online study conducted in the Netherlands exploring the feasibility of this novel approach. The results seem promising, but also highlight a number of methodological problems with this approach, most notably nontrading and the elicitation of negative values. Additional research is necessary to try to overcome these problems and to determine the potential of this new approach.

Comparison of Modes of Administration and Alternative Formats for Eliciting Societal Preferences for Burden of Illness.

BACKGROUND: Proposals for value-based assessment, made by the National Institute of Health and Care Excellence (NICE) in the UK, recommended that burden of illness (BOI) should be used to weight QALY gain. This paper explores some of the methodological issues in eliciting societal preferences for BOI. AIMS: This study explores the impact of mode of administration and framing in a survey for eliciting societal preferences for BOI. METHODS: A pairwise comparison survey with six arms was conducted online and via face-to-face interviews, involving two different wordings of questions and the inclusion/exclusion of pictures. Respondents were asked which of two patient groups they thought a publically funded health service should treat, where the groups varied by life expectancy without treatment, health-related quality of life (HRQOL) without treatment, survival gain from treatment, and HRQOL gain from treatment. Responses across different modes of administration, wording and use of pictures were compared using chi-squared tests and probit regression analysis controlling for respondent socio-demographic characteristics. RESULTS: The sample contained 371 respondents: 69 were interviewed and 302 completed the questionnaire online. There were some differences in socio-demographic characteristics across the online and interview samples. Online respondents were less likely to choose the group with higher BOI and more likely to treat those with a higher QALY gain, but there were no statistically significant differences by wording or the inclusion of pictures for the majority of questions. Regression analysis confirmed these results. Respondents chose to treat the group with larger treatment gain, but there was little support for treating the group with higher BOI. Respondents also preferred to treat the group with treatment gains in life expectancy rather than HRQOL. CONCLUSIONS: Mode of administration did impact on responses, whereas question wording and pictures did not impact on responses, even after controlling for the socio-demographic characteristics of respondents in the regression analysis.