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• Cost-effectiveness criteria often need to be relaxed for reimbursing orphan drugs. • We find that a large part of the public likely supports such differential treatment. • Support depends in part on patients' age, disease severity, and their health gains. • A considerable minority does not support differential treatment of orphan drugs. • Moral arguments regarding uniform decision-making often underlie lack of support. Orphan drugs, for patients with a rare disease, are increasingly available but often do not meet standard cost-effectiveness criteria for reimbursement. Consequently, policymakers regularly face the dilemma whether to relax these criteria for reimbursing orphan drugs. We examined whether—and why—there would be societal support for such differential treatment of orphan drugs. We conducted a labelled discrete choice experiment in a sample of the adult population (n = 1,172) in the Netherlands. Respondents were presented with ten choices on whether to reimburse an orphan drug given that a non-orphan drug with similar characteristics would not be reimbursed, because it was not cost-effective, and asked to explain their choices. We used random-intercept logit regression models and inductive coding for analysing the quantitative and qualitative data. Of the respondents, 36.4% consistently chose for reimbursing the orphan drug, mostly because "everyone is entitled to live a healthy life and good quality healthcare", and 17.3% consistently for not reimbursing the orphan drug, mostly because "[this] is unfair to patients with a common disease". The remaining 46.3% made alternating choices and were more likely to choose for reimbursing orphan drugs when patients were aged between 1 and 70 years, had moderate disease severity, and considerable health gain from treatment. This study finds considerable support but also strong preference heterogeneity amongst members of the public in the Netherlands for differential treatment of orphan drugs in reimbursement decisions, when these drugs do not meet common cost-effectiveness criteria. However, a substantial minority opposes differential treatment, mostly on moral grounds. [ABSTRACT FROM AUTHOR]

Objectives: Decision-making frameworks that draw on economic evaluations increasingly use equity weights to facilitate a more equitable and fair allocation of healthcare resources. These weights can be attached to health gains or reflected in the monetary threshold against which the incremental cost-effectiveness ratios of (new) health technologies are evaluated. Currently applied weights are based on different definitions of disease severity and do not account for age-related preferences in society. However, age has been shown to be an important equity-relevant characteristic. This study examines the willingness to pay (WTP) for health-related quality of life (QOL) gains in relation to the disease severity and age of patients, and the outcome of the disease.Methods: We obtained WTP estimates by applying contingent-valuation tasks in a representative sample of the public in The Netherlands (n = 2023). We applied random-effects generalized least squares regression models to estimate the effect of patients' disease severity and age, size of QOL gains, disease outcome (full recovery/death 1 year after falling ill), and respondent characteristics on the WTP.Results: Respondents' WTP was higher for more severely ill and younger patients and for larger-sized QOL gains, but lower for patients who died. However, the relations were nonlinear and context dependent. Respondents with a lower age, who were male, had a higher household income, and a higher QOL stated a higher WTP for QOL gains.Conclusions: Our results suggest that-if the aim is to align resource-allocation decisions in healthcare with societal preferences-currently applied equity weights do not suffice. [ABSTRACT FROM AUTHOR]

Background: Priority setting in healthcare can be guided by both efficiency and equity principles. The latter principle is often explicated in terms of disease severity and, for example, defined as absolute or proportional shortfall. These severity operationalizations do not explicitly consider patients' age, even though age may be inextricably related to severity and an equity-relevant characteristic.Objective: This study examines the relative strength of societal preferences for severity and age for informing allocation decisions in healthcare.Methods: We elicited preferences for severity and age in a representative sample of the public in The Netherlands (N = 1025) by applying choice tasks and person-trade-off tasks in a design in which severity levels and ages varied both separately and simultaneously between patient groups. We calculated person trade-off ratios and, in addition, applied ordinary least squares regression models to aid interpretation of the ratios when both severity and age varied.Results: Respondents attached a higher weight (median of ratios: 2.46-3.50) to reimbursing treatment for relatively more severely ill and younger patients when preferences for both were elicited separately. When preferences were elicited simultaneously, respondents attached a higher weight (median of ratios: 1.98 and 2.42) to reimbursing treatment for relatively younger patients, irrespective of patients' severity levels. Ratios varied depending on severity level and age and were generally higher when the difference in severity and age was larger between groups.Conclusions: Our results suggest that severity operationalizations and equity weights based on severity alone may not align with societal preferences. Adjusting decision-making frameworks to reflect age-related societal preferences should be considered. [ABSTRACT FROM AUTHOR]

• There was little evidence of socially desirable responding. • Cheap talk mitigation slightly improved external validity but not internal validity. • Research into other mitigation methods and sources of hypothetical bias is advised. Discrete choice experiments (DCEs) have been used extensively to elicit preferences. However, the hypothetical nature of choices induces socially desirable behaviour and endangers internal and external validity of DCEs. This study experimentally investigated social desirability bias in DCEs and whether it can be mitigated using the cheap talk mitigation method in the context of food choices. Respondents (N = 1027) were randomly allocated to one of four questionnaire versions: default without manipulation, priming socially desirable behaviour, cheap talk mitigation, or both. The effect on internal validity was assessed by comparing respondent-reported characteristics, DCE results, and prediction accuracy for a holdout task between questionnaire versions. The effect on external validity was assessed by comparing stated and revealed preferences. Social desirability bias, if present, was hardly affected by cheap talk mitigation. Respondent-reported characteristics, DCE results and prediction accuracy for the holdout task and actual food choice did not strongly differ between questionnaire versions. Prediction accuracy for the holdout task was lowest in the default version. Prediction accuracy for actual food choice was slightly better among respondents in the versions that were exposed to cheap talk mitigation. Social desirability bias was hard to detect and mitigate in this study, potentially due to limited social desirability, the effectiveness of the cheap talk mitigation method, and other sources of hypothetical bias. The differences in prediction accuracy indicates that cheap talk mitigation slightly improved external validity at minimum cost to internal validity. Recommendations for future research are provided. [ABSTRACT FROM AUTHOR]

Criteria used by the National Institute for Health and Care Excellence (NICE) to assess life-extending, end-of-life (EoL) treatments imply that health gains from such treatments are valued more than other health gains. Despite claims that the policy is supported by societal values, evidence from preference elicitation studies is mixed and in-depth research has shown there are different societal viewpoints. Few studies elicit preferences for policies directly or combine different approaches to understand preferences. Survey questions were designed to investigate support for NICE EoL guidance at national and regional levels. These ‘Decision Rule’ and ‘Treatment Choice’ questions were administered to an online sample of 1496 UK respondents in May 2014. The same respondents answered questions designed to elicit their agreement with three viewpoints (previously identified and described) in relation to provision of EoL treatments for terminally ill patients. We report the findings of these choice questions and examine how they relate to each other and respondents' viewpoints. The Decision Rule questions described three policies: DA – a standard ‘value for money’ test, applied to all health technologies; DB – giving special consideration to all treatments for terminal illnesses; and DC – giving special consideration to specific categories of treatments for terminal illnesses e.g. life extension (as in NICE EoL guidance) or those that improve quality-of-life (QoL). Three Treatment Choices were presented: TA – improving QoL for patients with a non-terminal illness; TB – extending life for EoL patients; and TC – improving QoL at the EoL. DC received most support (45%) with most respondents giving special consideration to EoL only when treatments improved QoL. The most commonly preferred treatment choices were TA (51%) and TC (43%). Overall, this study challenges claims about public support for NICE's EoL guidance and the focus on life extension at EoL and substantiates existing evidence of plurality in societal values. [ABSTRACT FROM AUTHOR]

Experiencing deteriorating health has implications for your quality of life. The theory of adaptation suggests that with time spend living in a health state individuals can adapt, resulting in observed quality of life levels to revert or stagnate despite persistently decreased health. Adaptation has implications for the use of subjective quality of life indicators when quantifying the impact of health changes or the benefits from new medical technologies. As both the impact from ill health and the benefit from new interventions might be disease- or subgroup-specific adaptation further raises ethical concerns but empirical evidence on its existence, magnitude, and heterogeneity remains inconclusive. This paper uses a general population sample of 9,543 individuals that participate in the UK Understanding Society survey and experience the onset of a long-standing illness or disability to provide evidence on these questions. Using ordered-response fixed effects models we explore longitudinal changes in self-assessed health and life satisfaction around the onset of disability. Our results indicate that disability onset is associated with large decreases in subjective health and well-being. Over time this initial decrease in subjective quality of life indicators attenuates, especially in life satisfaction and to a lesser extent for self-assessed health. While the relative difference in adaptation across these two measures remains persistent, we find that across demographic and severity groups the initial impact of disability onset and adaptation differs considerably in its magnitude. These results have important implications for studies aiming to quantify the impact of health conditions on quality of life outcomes, especially when using observational datasets. • Adaptation is particularly pronounced in life satisfaction. • Across subgroups adaptation in life satisfaction differs. • In self-assessed health adaptation is limited with no differences across subgroups. • When using well-being measures adaptation could distort measured disease impact. [ABSTRACT FROM AUTHOR]

Background Conditional reimbursement of new health technologies is increasingly considered as a useful policy instrument. It allows gathering more robust evidence regarding effectiveness and cost-effectiveness of new technologies without delaying market access. Nevertheless, the literature suggests that ending reimbursement and provision of a technology when it proves not to be effective or cost-effective in practice may be difficult. Objectives To investigate how policymakers and the general public in the Netherlands value removing a previously reimbursed treatment from the basic benefits package relative to not including a new treatment. Methods To investigate this issue, we used discrete-choice experiments. Mixed multinomial logit models were used to analyze the data. Compensating variation values and changes in probability of acceptance were calculated for withdrawal of reimbursement. Results The results show that, ceteris paribus, both the general public (n = 1169) and policymakers (n = 90) prefer a treatment that is presently reimbursed over one that is presently not yet reimbursed. Conclusions Apparently, ending reimbursement is more difficult than not starting reimbursement in the first place, both for policymakers and for the public. Loss aversion is one of the possible explanations for this result. Policymakers in health care need to be aware of this effect before engaging in conditional reimbursement schemes. [ABSTRACT FROM AUTHOR]

Objectives Recent debates in the Netherlands on health care priority setting have focused on the relative value of gains generated by life-extending medicines for people with a terminal illness, mostly new cancer drugs. These treatments are generally expensive, provide relatively small health gains, and therefore usually do not meet common cost per QALY thresholds. Nevertheless, these drugs may be provided under the assumption that there is public support for making a special case for treatments for people with a terminal illness. This study investigated the views of the public in the Netherlands on a range of equity and efficiency considerations relevant to priority setting and examines whether there is public support for making such a special case. Methods Using Q methodology, three viewpoints on important principles for priority setting were identified. Data were collected through ranking exercises conducted by 46 members of the general public in the Netherlands, including 11 respondents with personal experience with cancer. Results Viewpoint 1 emphasized that people have equal rights to healthcare and opposed priority setting on any ground. Viewpoint 2 emphasized that the care for terminal patients should at all times respect the patients’ quality of life, which sometimes means refraining from invasive treatments. Viewpoint 3 had a strong focus on effective and efficient care and had no moral objection against priority setting under certain circumstances. Conclusions Overall, we found little public support for the assumption that health gains in terminally ill patients are more valuable than those in other patients. This implies that the assumption that society is prepared to pay more for health gains in people who have only a short period of lifetime left does not correspond with societal preferences in the Netherlands. [ABSTRACT FROM AUTHOR]

Governments across Europe are required to make decisions about how best to allocate scarce health care resources. There are legitimate arguments for eliciting societal vales in relation to health care resource allocation given the roles of the general public as payers and potential patients. However, relatively little is known about the views of the general public on general principles which could guide these decisions. In this paper we present five societal viewpoints on principles for health care resources allocation and develop a new approach, Q2S, designed to investigate the extent to which these views are held across a range of European countries. An online survey was developed, based on a previously completed study Q methodology, and delivered between November 2009 and February 2010 across nine countries to 33,515 respondents. The largest proportion of our respondents (44%), were found to most associate themselves with an egalitarian perspective. Differences in views were more strongly associated with countries than with socio-demographic characteristics. These results provide information which could be useful for decision makers in understanding the pluralistic context in which they are making health care resource allocation decisions and how different groups in society may respond to such decisions. [ABSTRACT FROM AUTHOR]

Resources available to the health care sector are finite and typically insufficient to fulfil all the demands for health care in the population. Decisions must be made about which treatments to provide. Relatively little is known about the views of the general public regarding the principles that should guide such decisions. We present the findings of a Q methodology study designed to elicit the shared views in the general public across ten countries regarding the appropriate principles for prioritising health care resources. In 2010, 294 respondents rank ordered a set of cards and the results of these were subject to by-person factor analysis to identify common patterns in sorting. Five distinct viewpoints were identified, (I) “Egalitarianism, entitlement and equality of access”; (II) “Severity and the magnitude of health gains”; (III) “Fair innings, young people and maximising health benefits”; (IV) “The intrinsic value of life and healthy living”; (V) “Quality of life is more important than simply staying alive”. Given the plurality of views on the principles for health care priority setting, no single equity principle can be used to underpin health care priority setting. Hence, the process of decision making becomes more important, in which, arguably, these multiple perspectives in society should be somehow reflected. [ABSTRACT FROM AUTHOR]

The recent White Paper emphasises once more that the Trans-European transport network (TEN) is a key element in European Union (EU) policy, yet that its realisation is jeopardised by the lack of an integrated approach during evaluation and funding of cross-border infrastructure. Despite the intervention of the EU in conceptualising the TEN, subsidiarity and continued use of conventional evaluation procedures mean that most assessment of individual TEN components is still undertaken in relatively narrow, national terms. The primary objective of this paper is to explore how the international character of the TEN may be recognised and incorporated in still predominantly national evaluation processes, and which evaluation horizons may be broadened based on the concepts of network effects and European value added (EVA). We present some examples of EVA and discuss EU involvement in projects with potentially high EVA, either by co-ordinating bilateral negotiations for compensation between Member States or by co-financing projects that support the achievement of strategic EU transport policy objectives. [Copyright &y& Elsevier]

Health gains are increasingly weighted in economic evaluations of new health technologies to guide resource-allocation decisions in healthcare. In Norway and the Netherlands weights are, for example, based on the disease severity of patients. In England and Wales, a higher weight is attached to quality-adjusted life-years (QALYs) gained from life-extending end-of-life (EOL) treatments. Societal preferences for QALY gains in EOL patients are increasingly examined. Although the available evidence suggests that gains in health-related quality of life (QOL) may be preferred to gains in life expectancy (LE), little is known about the influence of EOL patients' age on these preferences. In this study, we examine the willingness to pay (WTP) for QOL and LE gains in EOL patients of different ages in a sample (n = 803) of the general public in the Netherlands. We found that WTP was relatively higher for QOL and LE gains in younger EOL patients. We further found indications suggesting that WTP may be relatively higher for QOL gains at the EOL, except for patients aged 20 for whom we observed a higher WTP for LE gains. Our results may inform discussions on attaching differential weights to QOL and LE gains in EOL patients of different ages with the objective to better align resource-allocation decisions with societal preferences. • Health gains in end-of-life (EOL) patients may receive a higher weight in decisions. • We examine the impact of patients' age and type of health gain on EOL preferences. • People are willing to pay more for health gains in younger EOL patients. • Quality of life gains are preferred over life expectancy gains at almost all ages. • Our results may help to refine EOL considerations in resource-allocation decisions. [ABSTRACT FROM AUTHOR]

To appropriately weight dimensions of quality of life instruments for health economic evaluations, population and patient preferences need to be elicited. Two commonly used elicitation methods for this purpose are discrete choice experiments (DCE) and case 2 best-worst scaling (BWS). These methods differ in terms of their cognitive burden, which is especially relevant when eliciting preferences among older people. Using a randomised experiment with respondents from an online panel, this paper examines the cognitive burden associated with colour-coded and level overlapped DCE, colour-coded BWS, and 'standard' BWS choice tasks in a complex health state valuation setting. Our sample included 469 individuals aged 65 and above. Based on both revealed and stated cognitive burden, we found that the DCE tasks were less cognitively burdensome than case 2 BWS. Colour coding case 2 BWS cannot be recommended as its effect on cognitive burden was less clear and the colour coding lead to undesired choice heuristics. Our results have implications for future health state valuations of complex quality of life instruments and at least serve as an example of assessing cognitive burden associated with different types of choice experiments. [ABSTRACT FROM AUTHOR]

There is a need for comprehensive measures to evaluate the benefits of health and social care services for older people. The newly developed Well-being of Older People measure (WOOP) aims to capture all aspects that older people find important to their well-being. This study explores the validity and test-retest reliability of the WOOP. Between December 2017 and January 2018, an online survey was used to retrieve data from 1113 people aged 65 years and older in the Netherlands. Regression analyses on Cantril's Ladder scores were conducted to explore the relative importance of the items of the WOOP. Dimensionality was checked using exploratory factor analysis. Convergent and discriminant validity were investigated by relating the WOOP to several measures of health and well-being. Test-retest reliability was examined using data from 269 respondents that participated in a second online survey, distributed one week after the first. The items of the WOOP were significantly associated with Cantril's Ladder scores. When regressed simultaneously this was still true for all but the 'social contacts' item and one level of the 'acceptance and resilience' item. The dimensionality analysis revealed three factors, of which two included items of the WOOP and the EQ-5D-5L and the third only items of the WOOP. The WOOP correlated moderately to highly with physical health, and (very) highly with (mental) health and well-being measures. The test-retest reliability in terms of ICC was high, whereas the kappa for the items was fair to good, except for two items. Overall, the WOOP seems to capture aspects relevant to the well-being of older people adequately, and the results of first validity and reliability tests were satisfactory. Before the WOOP can be used in economic evaluations, further validation in a variety of health and social care settings is recommended, and utility weights need to be determined. • First quantitative validation of the Well-being of Older People measure (WOOP). • Which captures a comprehensive set of well-being domains relevant to older people. • Validity and test-retest reliability were satisfactory among Dutch older people. • The WOOP appears to be a promising outcome measure for use in economic evaluations. [ABSTRACT FROM AUTHOR]

Economic evaluations of interventions in health and social care require outcome measures that capture their full benefits, including those beyond health. This study aimed to assess construct validity, test-retest reliability, and responsiveness of the newly developed 10-item Well-being instrument (WiX). Data were gathered via an online survey in a representative sample of the adult general population in The Netherlands (N = 1045). Construct validity was assessed by inspecting convergent, structural, and discriminant validity, following the COnsensus-based Standards for the selection of health status Measurement INstruments methodology. Regression analyses of the WiX and its items on other validated measures of well-being were performed to assess the convergent validity of the instrument and the relevance of its items. Dimensionality of the WiX was assessed using exploratory factor analysis. To assess discriminant validity, several hypotheses in terms of well-being differences were assessed. Finally, a second survey was sent out 2 weeks after the initial survey (n = 563; 53.9% response rate) to assess the test-retest reliability and responsiveness of the WiX. The WiX showed to be correlated with alternative well-being measures as expected and able to sufficiently differentiate between relevant subgroups in the population. Moreover, the dimensionality analysis indicated that the WiX captures a broad array of elements relevant to well-being, including physical and mental health. The test-retest reliability was good, with an intraclass correlation coefficient of 0.82. The results regarding the WiX are favorable and indicate that this new instrument may be a promising alternative for existing measures of well-being for evaluating interventions in health and social care. • Economic evaluations of interventions in health and social care require outcome measures that capture their full benefits, including those beyond health. For this purpose, outcome measures capturing overall well-being comprehensively are needed. • The 10-item Well-being instrument (WiX) was developed to improve welfare economic evaluations in the field of health and social care, while also allowing comparisons with interventions in other sectors or across sectors. The WiX comprehensively measures the satisfaction of members of the adult population on 10 important domains of well-being. • The WiX seems to be a promising alternative for existing measures of well-being for evaluating interventions in health and social care. [ABSTRACT FROM AUTHOR]

Background: Integrating patient preferences in Health Technology Assessment (HTA) is argued to improve uptake, adherence, and patient satisfaction. However, how to elicit and incorporate these preferences in HTA in a systematic and scientifically valid manner is subject to debate.Objective: This article provides a systematic review of the challenges to integrating patient preferences in HTA that have been raised in the literature about patient preferences in HTA.Methods: A systematic review of articles published between 2013 and 2017 addressing challenges to the integration of patient preferences in HTA was conducted in 7 databases. All issues with respect to the integration of patient preferences in HTA were extracted and divided into 5 categories: conceptual, normative, procedural, methodological, and practical issues. The issues were ranked according to how often they were mentioned.Results: Of 2147 retrieved articles, 67 were included in the analysis. Thirty-seven unique research issues were identified. In the majority of the articles, methodological issues were posed (82%), followed by procedural (73%), normative (51%), practical (24%), and conceptual (9%) issues. Frequently posed methodological issues concerned preference heterogeneity and choice of method. Common procedural issues concerned how to evaluate the impact of preference studies and their degree of being evidence based.Conclusions: This article provides an overview of issues with respect to the integration of patient preferences in HTA procedures. Most issues were of a methodological or procedural nature; yet, the large number of different issues points to the overall importance of further researching the different aspects concerned with patient preferences in HTA. Through its ranking of how many articles mention particular issues, this article proposes an implicit research agenda. [ABSTRACT FROM AUTHOR]

Objectives: Lost unpaid work and leisure time of patients due to ill health often are not included in economic evaluations, even in those taking a societal perspective. This study investigated the monetary value of unpaid work and leisure time to enable the inclusion of patient time in economic evaluations.Methods: A contingent valuation study was performed to derive monetary values of unpaid work and leisure time. Data were collected with an online survey among a representative sample of people 18 years and older in the Netherlands in terms of age, sex, and educational level in January 2014 (n = 316). Willingness-to-accept (WTA) and willingness-to-pay (WTP) values were analyzed with a two-part model. First, a logistic regression model investigated the willingness to trade in the WTA/WTP tasks. Second, a log-transformed ordinary least squares regression model analyzed the level of positive WTA and WTP values.Results: The average WTA value for unpaid work was €15.83, and the average WTA value for leisure time was €15.86. The mean WTP value for leisure time was €9.37 when traded against unpaid work, and €9.56 when traded against paid work. Differences in monetary values of unpaid work and leisure time were partly explained by respondents' income, educational level, age, and household composition.Conclusions: Researchers can adhere to the societal perspective by also including the value of hours of lost unpaid work and leisure time in economic evaluations. As a first indication of its value, we suggest applying the WTA value of €16. [ABSTRACT FROM AUTHOR]

• COVID-19 vaccine hesitancy was heterogeneous both in terms of time and magnitude. • Following COVID-19 news, trust levels, risk preferences, and vaccine confidence predicted intention reversals. • Lowering the cost of following the news for the population may raise vaccine uptake. This paper investigates the prevalence and determinants of three main states of people's willingness to be vaccinated (WTBV) against COVID-19 – willing, unwilling and hesitant – and the occurrence and predictors of shifts between these states over time. Understanding the dynamics of vaccine intentions is crucial for developing targeted campaigns to increase uptake and emergency response preparedness. A panel survey consisting of 9 quarterly waves of data collected between April 2020 and January 2022. Baseline data included 24 952 adults from Germany, UK, Denmark, the Netherlands, France, Portugal, and Italy recruited from online panels to construct census-matched nationally representative samples. Self-reported COVID-19 vaccine intention was the main outcome. Multinomial logit random effects models were used to analyze the relationships of interest. All results reported as relative risk ratios (RRR). Hesitancy to get vaccinated was the most unstable vaccine intention, with on average 42% of ever hesitant respondents remaining in this state through future waves, followed by the 'unwilling' (53%) and 'willing (82%). Following COVID-19 news, trust in information from the government, GPs and the WHO, risk preferences, risk perceptions, and confidence in vaccines (or lack thereof) predicted vaccination intention reversals. Risk preferences acted both as an impediment and as a facilitator for the vaccine uptake depending on the initial vaccine intention. This study revealed the dynamic nature of COVID-19 vaccine intentions and its predictors in 7 European countries. The findings provide insights to policymakers for designing more effective communication strategies, particularly targeted at hesitant and unwilling to vaccinate population groups, to increase vaccine uptake for future public health emergencies. [ABSTRACT FROM AUTHOR]

Policy makers need to make decisions regarding the allocation of scarce healthcare resources. We study preferences for investment in additional healthcare capacity and allocation between two regions, focusing on reducing waiting time for elective surgery for a physical health problem. We elicit preferences from a societal and an individual perspective, with unequal initial waiting times between the two regions. In an online survey, 1039 respondents were randomly assigned to one of three versions of the experiment: (1) a social planner perspective, placing respondents in the role of a policy maker; (2) an individual perspective where the respondent's own region was better off regarding initial waiting times; (3) an individual perspective where the individual's own region was worse off regarding initial waiting times. Respondents were asked to rank the status quo and five scenarios where the investment in additional capacity led to different distributions of shorter waiting times between regions. For all allocations we presented both the reduction in waiting time and the resulting final waiting time for both regions. We find that in version 1 of the experiment, preferences were in line with inequality aversion and Rawlsian preferences regarding final waiting time. In version 3, similar preferences were found, although here they also align with individualistic preferences. In version 2, preferences were more heterogeneous, with both individualistic and egalitarian preferences present. Concluding, individualistic and egalitarian preferences mostly concerned final waiting time. We therefore recommend policy makers to focus on the effect on final waiting time instead of the reduction of waiting time. • We studied preferences for investment and allocation of extra healthcare capacity. • From societal planner perspective, inequality aversion and worse-off were prioritized. • From individual better-off perspective, selfish and egalitarian preferences relevant. • From individual worse-off perspective, selfish preferences were relatively stronger. • In all perspectives, final waiting times more important than waiting time reductions. [ABSTRACT FROM AUTHOR]

Objectives: The COVID-19 pandemic forms an unprecedented public health, economic, and social crisis. Uptake of vaccination is critical for controlling the pandemic. Nevertheless, vaccination hesitancy is considerable, requiring policies to promote uptake. We investigate Dutch citizens' preferences for policies that aim to promote vaccination through facilitating choice of vaccination, profiling it as the norm, making vaccination more attractive through rewards, or punishing people who reject vaccination.Methods: We conducted a discrete choice experiment in which 747 respondents were asked to choose between policies to promote vaccination uptake and their impacts on the number of deaths, people with permanent health problems, households with income loss, and a tax increase.Results: Respondents generally had a negative preference for policies that promote vaccination. They particularly disliked policies that punish those who reject the vaccine and were more favorable toward policies that reward vaccination, such as awarding additional rights to vaccinated individuals through vaccination passports. Respondents who reject vaccination were in general much more negative about the policy options than respondents who consider accepting the vaccine. Nevertheless, vaccination passports are supported by both respondents who accept the vaccine, those who reject vaccination, and those who are unsure about vaccination.Conclusions: This study provides concrete directions for governments attempting to increase the vaccination uptake in ways that are supported by the public. Our results could encourage policy makers to focus on policy options that make vaccination easier and reward people who take the vaccine, as especially the implementation of vaccination passports was supported. [ABSTRACT FROM AUTHOR]

Active disinvestment of healthcare interventions (i.e. discontinuing reimbursement by means of a policy decision) has received limited public support in the past. Previous research has identified four viewpoints on active disinvestment among citizens in the Netherlands. However, it remained unclear how strong these viewpoints are supported by society, and by whom. Therefore, the current study aimed to 1) measure the support for these four viewpoints and 2) assess whether support is associated with background characteristics of citizens. In an online survey, a representative sample of adult citizens in the Netherlands (n = 1794) was asked to rate their agreement with short narratives of the four viewpoints on a 7-point Likert scale. The survey also included questions on sociodemographic characteristics, health status, healthcare utilization, and opinions about responsibility and costs in the healthcare context. Logistic regression models were estimated for each viewpoint to assess the association between viewpoint support and these characteristics. The support for the different viewpoints varied between 46.8% and 57.7% of the sample. Viewpoint support was associated with participants' age, gender, educational level, financial situation, healthcare utilization, opinion on the responsibility of the government for the health of citizens, and opinion on whether the increase in healthcare expenditure and health insurance premiums is considered a problem. Resistance to active disinvestment may partially be explained by the consequences of disinvestment citizens anticipate experiencing themselves. Citizens considering the increase in healthcare expenditure a larger problem were more supportive of disinvestment than those considering it less of a problem. • Four distinct viewpoints on disinvestment were supported by half of Dutch citizens. • Concerns on negative consequences of disinvestment are associated with resistance. • Concerns on rising healthcare costs are associated with support for disinvestment. [ABSTRACT FROM AUTHOR]

Objectives: This article reports the results of the application of conjoint analysis (CA) to derive a monetary value of informal care. Compared with normally recommended valuation methods such as the opportunity cost method and proxy good method, a valuation elicited through a CA might be more sensitive to the heterogeneity and dynamics of informal care. Methods: We developed a questionnaire and conducted a survey in which informal caregivers were asked to rate four different hypothetical informal caregiving situations (reflecting different combinations of care hours, care tasks, and monetary compensation). They were also asked to rate their current informal care situation compared with the four hypothetical situations. Data were obtained from postal questionnaires. These questionnaires were sent through regional support centres for informal caregivers of care recipients with various health problems. A total of 865 informal caregivers from this heterogeneous population returned a completed survey. Results: Informal caregivers require an increase of 81% in their hourly compensation for providing 21 instead of 7 hours informal care per week. This implies a compensation of &U20AC;12.36 per hour at a mean hypothetical compensation in the presented scenarios. We also found that an informal caregiver's current caregiving situation and other background characteristics were associated with the scenario ratings. Conclusions: We conclude that a CA is a promising alternative for existing methods to determine a monetary value of informal care and encourage more experiments in this area. [ABSTRACT FROM AUTHOR]

Abstract: This paper reports the results of the application of the opportunity costs and proxy good methods to determine a monetary value of informal care. We developed a survey in which we asked informal caregivers in The Netherlands to indicate the different types of time forgone (paid work, unpaid work and leisure) in order to be able to provide care. Moreover, we asked informal caregivers how much time they spent on a list of 16 informal care tasks during the week before the interview. Data were obtained from surveys in two different populations: informal caregivers and their care recipients with stroke and with rheumatoid arthritis (RA). A total of 218 care recipients with stroke and their primary informal caregivers completed a survey as well as 147 caregivers and their care recipients with RA. The measurement of care according to both methods is more problematic compared to the valuation. This is especially the case for the opportunity costs method and for the housework part in the proxy good method. More precise guidelines are necessary for the consistent application of both methods in order to ensure comparability of results and of economic evaluations of health care. [Copyright &y& Elsevier]

Objectives: Cost-effectiveness analyses typically require measurement of health-related quality of life (HRQoL) to estimate quality-adjusted life-years. Challenges with measuring HRQoL arise in the context of episodic conditions if patients are less likely-or even unable-to complete surveys when having disease symptoms. This article explored whether HRQoL measured at regular time intervals adequately reflects the HRQoL of people with epilepsy (PWE).Methods: Follow-up data from the Epilepsy Support Dog Evaluation study on the (cost-)effectiveness of seizure dogs were used in which HRQoL is measured in 25 PWE with the EQ-5D at baseline and every 3 months thereafter. Seizure count is recorded daily using a seizure diary. Regression models were employed to explore whether PWE were more likely to complete the HRQoL survey on a good day (ie, when seizures are absent or low in frequency compared with other days) and to provide an estimate of the impact of reporting HRQoL on a good day on EQ-5D utility scores.Results: A total of 111 HRQoL measurements were included in the analyses. Regression analyses indicated that the day of reporting HRQoL was associated with a lower seizure count (P<.05) and that a lower seizure count was associated with a higher EQ-5D utility score (P<.05).Conclusions: When HRQoL is measured at regular time intervals, PWE seem more likely to complete these surveys on good days. Consequently, HRQoL might be overestimated in this population. This could lead to underestimation of the effectiveness of treatment and to biased estimates of cost-effectiveness. [ABSTRACT FROM AUTHOR]

Background: Some studies in the Netherlands have gauged public views on principles for healthcare priority setting, but they fall short of comprehensively explaining the public disapproval of several recent reimbursement decisions.Objective: To obtain insight into citizens' preferences and identify the criteria they would propose for decisions pertaining to the benefits package of basic health insurance.Methods: Twenty-four Dutch citizens were selected for participation in a Citizen Forum, which involved 3 weekends. Deliberations took place in small groups and in plenary, guided by 2 moderators, on the basis of 8 preselected case studies, which participants later compared and prioritized under the premise that not all treatments can or need to be reimbursed. Participants received opportunities to inform themselves through written brochures and live interactions with 3 experts.Results: The Citizen Forum identified 16 criteria for inclusion or exclusion of treatments in the benefits package; they relate to the condition (2 criteria), treatment (11 criteria), and individual characteristics of those affected by the condition (3 criteria). In most case studies, it was a combination of criteria that determined whether or not participants favored inclusion of the treatment under consideration in the benefits package. Participants differed in their opinion about the relative importance of criteria, and they had difficulty in operationalizing and trading off criteria to provide a recommendation.Conclusions: Informed citizens are prepared to make and, to a certain extent, capable of making reasoned choices about the reimbursement of health services. They realize that choices are both necessary and possible. Broad public support and understanding for making tough choices regarding the benefits package of basic health insurance is not automatic: it requires an investment. [ABSTRACT FROM AUTHOR]

Based on theoretical notions, there is consensus that alternative payment models to the common fee-for-service model have the potential to improve healthcare quality through increased collaboration and reduced under- and overuse. This is particularly relevant for maternity care in the Netherlands because perinatal mortality rates are relatively high in comparison to other Western countries. Therefore, an experiment with bundled payments for maternity care was initiated in 2017. However, the uptake of this alternative payment model remains low, as also seen in other countries, and fee-for-service models prevail. A deeper understanding of stakeholders' perspectives on payment reform in maternity care is necessary to inform policy makers about the obstacles to implementing alternative payment models and potential ways forward. We conducted a Q-methodology study to explore perspectives of stakeholders (postpartum care managers, midwives, gynecologists, managers, health insurers) in maternity care in the Netherlands on payment reform. Participants were asked to rank a set of statements relevant to payment reform in maternity care and explain their ranking during an interview. Factor analysis was used to identify patterns in the rankings of statements. We identified three distinct perspectives on payment reform in maternity care. One general perspective, broadly supported within the sector, focusing mainly on outcomes, and two complementary perspectives, one focusing more on equality and one focusing more on collaboration. This study shows there is consensus among stakeholders in maternity care in the Netherlands that payment reform is required. However, stakeholders have different views on the purpose and desired design of the payment reform and set different conditions. Working towards payment reform in co-creation with all involved parties may improve the general attitude towards payment reform, may enhance the level of trust among stakeholders, and may contribute to a higher uptake in practice. • This study reveals three perspectives on payment reform in Dutch maternity care. • There is consensus across perspectives on the need for payment reform. • The perspectives differ in their focus on either outcomes, equality or collaboration. • Disagreement remains on the goal, conditions and desired design of the reform. • Co-creation from the start is crucial to facilitate trust and promote uptake of reform. [ABSTRACT FROM AUTHOR]

• Q methodology can be used to explore the perspectives of young children. • Two four-factor solutions were selected as comprehensive and coherent solution. • Factor differences highlight the importance of examining preferences individually. • A one-size-fits-all approach is not appropriate for addressing social needs. Not all children benefit from the opportunities of inclusive education, especially children with social, emotional and behavioural difficulties (SEBD). This article presents the findings of a Q study exploring the perspectives of children with or at risk of SEBD, who experience difficulties with their social participation within the mainstream classroom. Forty-five children, aged 6–8 years, sorted 15 statements outlining approaches for resolving social exclusion and victimisation situations. Four shared perspectives were identified per situation using by-person factor analysis. These perspectives differed primarily with respect to the actors held responsible for resolving the situation. Therefore, a one-size-fits-all approach is not appropriate for addressing the social needs of children with or at risk of SEBD within the inclusive classroom. [ABSTRACT FROM AUTHOR]

Objectives: To elicit willingness-to-accept (WTA) values for informal care based on the preferences of informal carers and noncarers. Methods: Two surveys were conducted with a sample of carers (n = 202) and a sample of noncarers (n = 200). Individuals in both groups were asked three questions in which they had to state the minimum monetary compensation they would require (WTA) if they had to look after a person described in a hypothetical scenario for one extra hour per day. Furthermore, carers were asked for the compensation they would demand if they had to be in charge of their actual care recipient for one extra hour per day. Results: No significant differences were found between the distributions of carers' and noncarers' WTA values. Overall, respondents' valuations were sensitive to and consistent with their preferences over the tasks to be carried out in the extra hour of informal care. On average, carers required a lower monetary compensation for one extra hour taking care of their loved one (mean/median WTA values €5.2/€4.5) than if they had to devote that time to look after the hypothetical care recipient (mean/median WTA values €6.4/€5.5). More than half of the carers stated the same value under the two caring situations, which suggests that carers' WTA values were influenced by their own experience providing informal care. Conclusions: Our results show that it is feasible to derive a monetary valuation for informal care from the preferences of noncarers. [ABSTRACT FROM AUTHOR]

Health economic evaluations using common health-related quality of life measures may fall short in adequately incorporating all relevant benefits of health and social care interventions targeted at older people. The Well-being of Older People measure (WOOP) is a broader well-being measure that comprises nine well-being domains. The objective of this study was to estimate a utility tariff for the WOOP, to facilitate its application in cost-utility analyses. A discrete choice experiment (DCE) with duration approach was set up and fielded among 2,012 individuals from the Netherlands aged 65 years and above. Matched pairwise choice tasks, colour-coding and level overlap were used to reduce the cognitive burden of the DCE. The choice tasks were created using a Bayesian heterogeneous D-efficient design. The estimation procedure accommodated for nonlinear time preferences via an exponential discounting function. The estimation results showed that 'physical health', 'mental health', and 'making ends meet' were the most important well-being domains for older people, followed by 'independence' and 'living situation'. Of somewhat lesser importance were domains like 'social life', 'receiving support' and 'feeling useful'. The generated utility tariffs can be used to translate well-being states described with the WOOP to a utility score between −0.616 and 1. This study established a tariff for the WOOP, which will facilitate its use in economic evaluations of health and social care interventions targeted at older people, first of all in the Netherlands. • A choice experiment was used to create weights for a novel well-being measure. • This enables the use of the WOOP instrument in economic evaluations of elderly care. • Health, independence, and financial stability were the most important dimensions. • Social contact, receiving support, or feeling useful were less important. • Study adopted methodological advances in utility measurement and choice task design. [ABSTRACT FROM AUTHOR]

Background and objective: Positive aspects of caring are commonly cited by carers and can potentially mediate carer strain. However, in outcome assessment for carers, it is still common practice to focus on the negative aspects of caring. This study explored the feasibility and validity of including positive items in a commonly used outcome measure—the Caregiver Strain Index (CSI). Methods: Five positive items were inserted into the CSI. The resulting ‘CSI+’ questionnaire was included in a postal survey of carers in the Netherlands. Feasibility was assessed through completion rates for the CSI+ and individual items. Validity was assessed through tests of the association of the positive items and the CSI+ scores with relevant characteristics of the care (construct validity) and related scales (convergent validity). Factor analysis was carried out the CSI+ scale. Results: The addition of the positive items did not noticeably affect the feasibility of the CSI, but did appear to improve the convergent validity of the measure. The multivariable analysis and factor analysis suggests that positive aspects of care could be grouped into ‘coping’ factors and ‘attitudinal’ factors. Conclusion: These findings support the use of positive items in outcome assessment for carers and provide some support for the use of the CSI+ as an instrument for doing this. [Copyright &y& Elsevier]

Vaccination is generally considered the most direct way to restoring normal life after the outbreak of COVID-19, but the available COVID-19 vaccines are simultaneously embraced and dismissed. Mapping factors for vaccine hesitancy may help the roll-out of COVID-19 vaccines and provide valuable insights for future pandemics. We investigate how characteristics of a COVID-19 vaccine affect the preferences of adult citizens in the Netherlands to take the vaccine directly, to refuse it outright, or to wait a few months and first look at the experiences of others. An online sample of 895 respondents participated between November 4th and November 10th, 2020 in a discrete choice experiment including the attributes: percentage of vaccinated individuals protected against COVID-19, month in which the vaccine would become available and the number of cases of mild and severe side effects. The data was analysed by means of panel mixed logit models. Respondents found it important that a safe and effective COVID-19 vaccine becomes available as soon as possible. However, the majority did not want to be the first in line and would rather wait for the experiences of others. The predicted uptake of a vaccine with the optimal combination of attributes was 87%, of whom 55% preferred to take the vaccine after a waiting period. This latter group tends to be lower-educated. Older respondents gave more weight to vaccine effectiveness than younger respondents. The willingness to take a COVID-19 vaccine is high among adults in the Netherlands, but a considerable proportion prefers to delay their decision to vaccinate until experiences of others are known. Offering this wait-and-see group the opportunity to accept the invitation at a later moment may stimulate vaccination uptake. Our results further suggest that vaccination campaigns targeted at older citizens should focus on the effectiveness of the vaccine. • We conducted a choice experiment on vaccination preferences among Dutch citizens. • 87% accepts a safe and effective COVID-19 vaccine that is available swiftly. • 55% of this group prefers to delay their decision to learn from others' experiences. • Respondents who preferred to wait tend to be lower-educated. • The vaccine's effectiveness is important for older respondents who prefer to wait. [ABSTRACT FROM AUTHOR]

This paper reports the results of the application of the conjoint measurement method (CM) to determine a monetary value of informal care. Compared to the normally recommended valuation methods, like the opportunity cost method and proxy good method, CM is probably better able to capture the heterogeneity of informal care. We developed a survey in which informal caregivers were asked to rate four different hypothetical informal caregiving situations, which differed with respect to care hours, care tasks and monetary compensation. Data were obtained from postal surveys. A total of 135 pairs of informal caregivers and care recipients with rheumatoid arthritis (RA) from the Netherlands returned a completed survey and were used in the analysis. Informal caregivers require an extra compensation of 1.00 euro per hour for providing one additional hour of the same informal care task (meaning that from the seventh to the eighth hour, they require 8 euro). For providing two extra hours of care, they require 2.00 euro compensation per hour. The relative valuation of informal care tasks is very diverse. Respondents require a compensation of 13.43 euro per hour for switching from providing light housework to personal care and 0.56 euro per hour for switching from providing personal care to heavy housework. Though CM is sometimes regarded as cognitively complex, 70% of the respondents were able and willing to evaluate the hypothetical caregiving scenarios. Elderly respondents especially had more difficulty with the method. In sum, CM is seen as a promising alternative for existing methods to determine a monetary value of informal care. The presented valuations of informal care can be incorporated in the numerator of a cost-effectiveness ratio in economic evaluations of health care. [ABSTRACT FROM AUTHOR]

Population ageing and restricted budgets result in the need for an efficient allocation of scarce resources in care services for older people. As these services tend to address more than only health, diverse wellbeing measures have been developed to assess their benefits in economic evaluations. These measures are grounded in research on wellbeing of older people and its determinants. Little is known about possible heterogeneity in this context and the extent to which wellbeing measures cover the aspects of wellbeing that are most important to older people with different views on wellbeing. We conducted a Q-methodology study between December 2016 and October 2017 to investigate the variety in views among people aged 65 and older in the Netherlands on what is important to their wellbeing. A purposive sample of 53 respondents ranked 34 opinion statements according to importance to their wellbeing and explained their ranking during a follow-up interview. Data were analysed using by-person factor analysis to identify common patterns in the rankings of the statements. Five distinct views were extracted in which different aspects were considered important: (I) health, financial security and a life partner; (II) family, support and physical functioning (III); autonomy, mental health and helping others; (IV) social contacts, support, mental health and religion; and (V) a life partner, social contacts, living environment and adaptation. This heterogeneity in views of older people on what constitutes wellbeing supports the use of person-centered approaches in care services for older people. Arguably, (evaluations of) policies and services for older people should take this plurality into consideration. • Little is known about the views of older people on what constitutes wellbeing. • This study found five distinct views on what they find important for wellbeing. • Each view accentuates specific domains or aspects within domains of wellbeing. • Only the domains health and social relations were important in most views. • Health and social care services for older people should consider this plurality. [ABSTRACT FROM AUTHOR]