Introduction
Material and methods
Search strategy
Selection of publications
Data extraction and process of synthesis
Synthesis
Results
First-level themes | Supporting quotes | Support in literature (citations in italic) |
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Patients better informed (know better what it is like) | “The argument for using patient values seems to hinge crucially on the fact that patients know the health states better than someone trying to imagine them. … Given the evidence that general population values are poor proxies for those of patients, this implies that patient values should be used.” “If the perspectives of patient and doctor are both valuable for their particular insights, it is less easy to see why this should also be true of the general public. Why should we think the public perspective important, if it lacks both experience and knowledge? It is ironic that current measures rely almost exclusively on eliciting the preferences of the public, when intuitively it seems to be the least authoritative group.” | Brazier (2005) p. 204 Wolff (2012) p. 460 Jalukar (1998) Revicki (1998) Gabriel (1999) Nord (1999) De Wit (2000) Ubel (2000) Polsky (2001) Happich (2002) Landy (2002) Menzel (2002) Feeny (2003) Prosser (2003) Stein (2003) Happich (2005) Burström (2006) Rashidi (2006) Ratcliffe (2007) Briggs (2008) Dolan (2008) Lloyd (2008) Stiggelbout (2008) Arnold (2009) Dolan (2009) Gandjour (2010) Garau (2011) Krabbe (2011) McTaggart-Cowan (2011a) McTaggart-Cowan (2011b) Stamuli (2011) McTaggart-Cowan (2012) Rand-Hendriksen (2012) Butt (2013) Burström (2014) Mulhern (2014) Thavorncharoensap (2014) Wang (2014) Whately-Smith (2014) Aronsson (2015) Gandhi (2015) Papageorgiou (2015) Rowen (2015) Schwalm (2015) Dagklis (2016) Jonker (2016) Mott (2016) Versteegh (2016) Gandhi (2017) Sossong (2017) |
The general public has an incomplete health state description | “First, the general public does not necessarily know what it is like to experience the specific illnesses being evaluated in CEAs, whereas patients actually experience the illnesses in question. (…) Second, when conducting utility elicitations from the general public, we must describe the health states in question to the public. These descriptions will always be incomplete and, therefore, may introduce bias.” | Ubel (2000) p 128 Ubel (2000) Happich (2005) Arnold (2009) McTaggart-Cowan (2011a) Schwalm (2015) Sossong (2017) |
The general public disregards adaptation | “Patients adapt to their illnesses, whereas people who have not experienced such a health state might fail to anticipate this ability to adapt.” | Edelaar-Peteers (2012) p. 806 Ubel (2000) Polsky (2001) Menzel (2002) Ubel (2003) Damschroder (2005) Happich (2005) Damschroder (2008) Dolan (2008) Stiggelbout (2008) Gandjour (2010) Garau (2011) McTaggart-Cowan (2011b) McTaggart-Cowan (2012) Wolff (2012) Burström (2014) Sun (2015) Jonker (2016) Versteegh (2016) Rowen (2017) |
Activity adjustment (as a form of adaptation) | “Realizing that a disease or disability is likely to be chronic, people may adjust their activities. Still desiring physical exercise, for example, a former cyclist, now paraplegic, may take up aerobic wheelchairing. Or a person may change occupations, not because she has altered her substantive goals in life, but because she now deems a different occupation to be a better avenue for achieving them.” | Menzel (2002) p. 2151 Versteegh (2016) |
Skill enhancement (as a form of adaptation) | “With time, chronically disabled or ill persons may develop greater skill in using whatever physical or mental capacities they retain. No activities or goals are adjusted; people simply improve their ability to accomplish their existing goals in their existing activities, beyond what they previously could ever have imagined was possible.” “…the authors note that ‘skill enhancement,’ ‘activity adjustment’ … could be considered ‘laudable adaptation’…” | Menzel (2002) p. 2151 Versteegh (2016) p. 70 McTaggart-Cowan (2011b) |
Substantive goal adjustment (as a form of adaptation) | “People may adjust not only the activities they select to pursue their goals, but the content and direction of the goals themselves. Their basic interest has changed.” | Menzel (2002) p. 2151 Versteegh (2016) |
Altered conception of health (as a form of adaptation) | “It is to note that people who have what is commonly thought of as ‘disability’ or ‘disease’ may be stimulated to adopt a radically different and, in their eyes, a more insightful definition of their health.” | Menzel (2002) p. 2152 McTaggart-Cowan (2011b) Versteegh (2016) |
Heightened stoicism (as a form of adaptation) | “They control their happiness so that it is a function only of what they come to see as achievable.” | Menzel (2002) p. 2152 |
Contrast effects (new, forgiving perspective on minor issues) | “For example, if a patient with MS is evaluating the probable quality of life of another hypothetical person with MS, she might have learned from her own experience that MS has made it easier for herself to emotionally deal with minor day-to-day frustrations that used to bother her significantly. (…) A member of the general public, on the other hand, may not consider the likelihood that having MS would create these types of contrast effects.” | Ubel (2003) p. 604 |
Focusing effects (the general public over-emphasize negative aspects of the health state, incl. Peak-start rule and Transition) | “[G]eneral population respondents typically focus on the negative aspects of ill health whilst ignoring unaffected life domains that the descriptive system does not bring to their attention…” “There are at least three factors that tend to inflate the public’s assessments of the severity of hypothetical health states. First … the respondent’s attention is drawn to the transition from one state to another. … Therefore, valuations are likely to be affected by a ‘Peak-Start Rule’ where respondents focus on the worst effects of a health change and the effects that are experienced immediately. For many adverse conditions, the peak and the start will coincide. …" | Garau (2011) p. 679 Dolan (2008) p. 71 Ubel (2003) Brazier (2005) McTaggart-Cowan (2011a) Peeters (2011) Stamuli (2011) Wolff (2012) Burström (2014) Wilson (2014) Sun (2015) Versteegh (2016) Leidl (2017) |
Reference point (distance renders evaluation more difficult) | “[F]or the general public impaired health may be too hypothetical, causing the general public to overestimate the impact of health impairments yielding rather low, and perhaps relatively often negative, that is "worse than dead", preferences …” | Versteegh (2016) p. 69 |
Valuation compression (distance compresses small differences between health states; the problem from the healthy viewpoint) | “"[T]he general population may undervalue movements between severe states. … [F]or example, at extreme levels of disability the general population may be insensitive to small improvements in mobility that are highly valued by patients.” “Much the way the distance we are from two objects affects our ability to judge the distance between them, people's current health affects their evaluation of severity of other health states.” … “To a person in normal health, the difference between hemiplegia and hemiplegia with aphasia may seem small - both health states are extremely severe. But, to a patient living with either health state, the difference will appear much larger - having or not having the ability to speak makes a big difference to these patients.” | Brazier (2005) p. 205 Ubel (2003) p. 602 Damschroder (2005) Garau (2011) |
Discrimination avoidance | “'Third, the public may be biased against people with disability or illness, and this may be reflected in value measurements.” | Ubel (2000) p. 128 Hadorn (1992) Lenert (1999) Brazier (2005) |
Protection of the unhealthy | “[T]he general population tend to give a lower health state value than do patients. (…) Giving lower values to the lives of ill people means that life-saving interventions will look less attractive than if patient values had been used…” “The ‘gain’ from an intervention [that is life extending] may then be higher when valued by patients” [i.e., when patients value health states higher than the general public].” | Brazier (2005) p. 205 Versteegh (2016) p. 70 Ubel (2000) McPherson (2004) |
Welfare economics | “An important normative position (or value judgement) in welfare economics is that the well-being of a society is simply the aggregation of the utility of individual members of society. In other words, it asserts the supremacy of an individual’s valuation of their own well-being. This implies that it is the preferences of the losers and gainers from a public programme that should be elicited, and not a sample of the general population who will be unaffected by the change; this would seem to suggest that patient values should be used.” “A strong argument in favor of using patient preferences in the assessment of health states is experience with a disease. What this study adds is a theoretical justification of patient preferences: they can be justified by Harsanyi’s veil-of-ignorance model and, more generally, by preference utilitarianism and welfare economics because their satisfaction increases or maximizes social welfare. … In contrast, this article found no compelling theoretical basis for community preferences for use in QALYs.” | Brazier (2005) p. 204 Gandjour (2010) p. E61 De Wit (2000) Burström (2006) Ratcliffe (2007) Brouwer (2008) Lee (2011) Stamuli (2011) Weyler (2011) Wang (2014) Schwalm (2015) Mott (2016) Gries (2016) Leidl (2017) |
Patients more affected | “The main rationale for using the patient’s perspective is to place greater emphasis on those most directly affected by a policy.” | McTaggart-Cowan (2011b), p. 1904 Froberg (1989) Dolan (1996) Gabriel (1999) Neumann (2000) McTaggart-Cowan (2011b) |

First-level themes | Supporting quotes | Support in literature (citations in italic) |
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Patients overestimate the quality of life of the ill health state (as a form of adaptation) | “Patients’ ability to adapt to illness might mean that they will overestimate the quality of life of specific health conditions.” | Ubel (2000) p. 130 Rand-Hendriksen (2012) Burström (2014) Ogorevc (2017) Rowen (2017) |
Happy slave (as a form of adaptation) | “The ‘happy slave’ example shows why subjective reports of well-being or the expression of someone's preferences might both be inadequate guides to value; even if a slave is happy, and prefers slavery to freedom, nevertheless, it is argued, he or she has real interests that are undermined by slavery." “From an external point of view one can have reason to value some conditions above others even if they are not correlated with greater subjective well-being. As in the 'happy slave' case, the existence of choice, freedom, and opportunity seems to matter.” | Wolff (2012) pp. 458, 460 Burström (2006) |
Cognitive denial (as a form of adaptation) | “Patients may find it difficult to admit how poor their objective, functional health really is. … [I]t hardly seems desirable to base the value of a health state that is used to shape social policy on judgments that are factually mistaken.” | Menzel (2002) p. 2151 Ubel (2000) McTaggart-Cowan (2011b) Versteegh (2016) |
Failure to recognize full health (as a form of adaptation) | “[P]atients cease to realize anymore what full health is like or what it would enable them to do. Here again, it hardly seems desirable for such cognitive blindness to influence the measurement of health state utility…” | Menzel (2002) p. 2151 Dolan (2008) McTaggart-Cowan (2011b) Jonker (2016) Versteegh (2016) Rowen (2017) |
Heightened stoicism | “They [patients] control their happiness so that it is a function only of what they come to see as achievable.” | Menzel (2002) p. 2152 |
Lowered expectations | “Regardless of other, more complex adjustments, chronically ill or disabled persons may simply lower their level of expected achievement, fatalistically accepting their diminished lot in life.” “Some researchers argue that certain elements of adaptation (such as the lowering of expectations) are regrettable and that it would be inappropriate for these factors to influence healthcare prioritization decisions.” | Mentzel (2002) p. 2152 Garau (2011) p. 679 Ubel (2000) Ratcliffe (2007) McTaggart-Cowan (2011b) Stamuli (2011) McTaggart-Cowan (2012) Jonker (2016) Versteegh (2016) Rowen (2017) |
Response shift | “Imagine a person who is asked before and after the onset of an illness to rate her HRQoL on a 1 to 10 scale. The illness may change her idea of what the numbers 1 and 10 represent, a phenomenon called response shift – changes in health lead to changing internal standards for evaluating one’s own health, making it difficult to compare HRQoL before and after illness. (…) Response shift is also related to changing expectations.” | Ubel (2003) p. 602 Myers (2003) Happich (2005) Finell (2012) McTaggart-Cowan (2012) |
Focusing effects due to patients’ recall difficulties (including peak-end-rule) | “There is substantial evidence that people are generally poor at recalling their experiences. People recalling past experiences tend to be subject to a range of biases, such as 'peak-end' effects where they tend to focus on their peak experience and their last experience and, consequently, weight these experiences more heavily than the rest.” “There is now good evidence that the retrospective recall of health is highly correlated with current health state and not so well correlated with the initial state. More generally, our memories do not recall past experiences and their duration particularly well.” | Brazier (2005) p. 206 Dolan (2008) p. 72 Ubel (2003) |
Reference point (distance render evaluation more difficult) | “The general public is generally healthy and can judge the loss of capabilities from the viewpoint of someone who is in full health. This may result in a 'better', or at least uniform, representation of the 'distance' between being in full health and having the health impairment. For patients the reference point they are reasoning from may have shifted and full health may be too hypothetical, which may be con-sidered problematic when wishing to come to universally applicable health state valuations.” | Versteegh (2016) p. 69 |
Valuation compression (distance compresses small differences between health states; problem from the patient viewpoint) | “When patient values are used in studies aimed at the comparison of different therapeutic modalities for one clinical problem, the ‘valuation compression’ at the upper end of the scale might result in loss of sensitivity to discriminate between the therapeutic modalities, when in fact differences between those modalities exist.” | de Wit (2000) p. 117 |
Societal perspective | “[V]aluations made by the general public may be most appropriate in situations where health care resources are being distributed in the public interest, such as in government funded health systems. Their appropriateness in these instances stems from the belief that valuations made by the general public are the best representation of societal preferences." “[T]he current economic standard is to elicit HRQoL estimates from the general public rather than from patients, because economic analyses are meant to guide social policy and not individual patient decisions.” “[E]ven if the [patients’] values derived would be unbiased in terms of costs, they may not necessarily mirror community preferences, and hence (…) may lack democratic legitimacy.” | Polsky (2001) p. 34 Ubel (2003) p. 604 Happich (2005) p. 51 Neumann (2000) Ubel (2000) Polsky (2001) Happich (2002) Myers (2003) Prosser (2003) McPherson (2004) Brazier (2005) Stein (2005) Ariza-Ariza (2006) Ratcliffe (2007) Dale (2008) Lloyd (2008) Stiggelbout (2008) Arnold (2009) Garau (2011) Lee (2011) McTaggart-Cowan (2011b) Stamuli (2011) Pickard (2013) Burström (2014) Wang (2014) Whately-Smith (2014) Gandhi (2015) Jonker (2016) Mott (2016) Versteegh (2016) Sossong (2017) |
Insurance (ex ante) | “Public funding can essentially be seen as public insurance and so it is the ex ante public preferences that should be used to value health states.” | Brazier (2005) p. 204 Ratcliffe (2007) Arnold (2009) Gandjour (2010) Burström (2014) Versteegh (2016) |
Tax payer (including insurance payer) | “A point often raised is that in a tax or insurance system of health, the people paying for health care are ordinary citizens, many of whom do not fall seriously ill. It might be claimed that those who pay have a right to determine how their money is spent.” “This decision [based on the Panel on Cost-effectiveness in Health and Medicine, to use values from the general public] was based on the argument that the public bears the costs associated with healthcare decisions, hence they should be part of the decision-making process for the allocation of benefits.” | Wolff (2012) p. 461 Stamuli (2011) p. 206 Froberg (1989) Hadorn (1992) Dolan (1996) de Wit (2000) Green (2003) Myers (2003) Brazier (2005) Burström (2006) Rashidi (2006) Ratcliffe (2007) Happich (2009) Gandjour (2010) McTaggart-Cowan (2011a) Rand-Hendriksen (2012) Butt (2013) Burström (2014) Mulhern (2014) Thavorncharoensap (2014) Wang (2014) Whately-Smith (2014) Aronsson (2015) Gandhi (2015) Rowen (2015) Schwalm (2015) Mott (2016) Versteegh (2016) Ogorevc (2017) Rowen (2017) |
Welfarism/Extra-welfarism | “It could be argued that every citizen in a public system has an option to use the service and so may be gainers or losers, meaning that the general population would be a good proxy." “… an ‘extra-welfarist’ approach, which considers that any number of stakeholders, such as social decision makers (…) or an average tax-payer, might be regarded as a more appropriate source of values than individual patients.” | Brazier (2005) p. 204 Stamuli (2011) p. 205 Arnold (2009) Gries (2016) |
Veil of ignorance | “'The Washington Panel went on to use the ‘veil of ignorance’ to support the use of community values, where a ‘rational public decides what is the best course of action when blind to its own self-interest, aggregating the utilities of persons who have not vested interest in particular health states seems most appropriate.’” “[I]t is stated that rational citizens when operating behind a ‘veil of ignorance’, and thus ignorant of their own future health state and needs, would prefer that societal decisions lead to maximum aggregate benefit within that society.” | Brazier (2005) p. 204 de Wit (2000) p. 110 Ubel (2000) Happich (2002) Rashidi (2006) Pyne (2009) McTaggart-Cowan (2011a) Gandjour (2010) Stamuli (2011) McTaggart-Cowan (2011b) Rand-Hendriksen (2012) Butt (2013) Thavorncharoensap (2014) Schwalm (2015) Mott (2016) Versteegh (2016) Ogorevc (2017) |
Vested interest | “A further argument is that unlike patients, the general population tends not to have a vested interest in getting access to treatment and is, therefore, more likely to give an unbiased view of the value of the health gain it generates.” | Garau (2011) p. 679 Ashby (1994) Jalukar (1998) Polsky (2001) Happich (2002) Happich (2005) Rashidi (2006) Briggs (2008) Pyne (2009) McTaggart-Cowan (2011a) McTaggart-Cowan (2011b) Stamuli (2011) McTaggart-Cowan (2012) Rand-Hendriksen (2012) Butt (2013) Whately-Smith (2014) Gandhi (2015) Papageorgiou (2015) Rowen (2015) Schwalm (2015) Dagklis (2016) Jonker (2016) Mott (2016) Rowen (2017) |
Better for patients that non-adapted health states are considered | “[I]t may be unjust if some patients lost out in the race for resources because their effort diminished the value of treatments for them compared to other patients who did not expend the same effort’. I.e. they have adapted…” | Dolan (2008) p. 75 Menzel (2002) McPherson (2004) |
Better for patients with general public values: the general public rates conditions as worse; gains from treatment hence appear larger | “It has been argued that the use of general population values benefits patients. This is based on the observation that the general population tends to give a lower health state value than do patients. Therefore, for any intervention aimed at curing or preventing a condition associated with ill health states, general population values will generate a larger gain.” | Brazier (2005) p. 205 Raisch (2000) Versteegh (2016) Ogorevc (2017) |
Easier to obtain large amounts of data | “One of the major perceived advantages of such generic measures of health status [letting representatives of the general population evaluate descriptions of health conditions] from the researcher’s perspective lies in their ability to provide ‘off the shelf’ values for a wide variety of generic health states.” | Ratcliffe (2007) p. 396 Gandjour (2010) Rand-Hendriksen (2012) Wolff (2012) Rowen (2015) Mott (2016) Ogorevc (2017) |

Discussion
No flawless position
An important normative position (or value judgement) in welfare economics is that the well-being of a society is simply the aggregation of the utility of individual members of society. In other words, it asserts the supremacy of an individual’s valuation of their own well-being. This implies that it is the preferences of the losers and gainers from a public programme that should be elicited, and not a sample of the general population who will be unaffected by the change; this would seem to suggest that patient values should be used. One response to this is that current patients do not represent all those likely to be affected by the set of decisions being made. It could be argued that every citizen in a public system has an option to use the service and so may be gainers and losers, meaning that the general population would be a good proxy. However, this presumes that all citizens have an equal chance of receiving all forms of care, which is unlikely. Therefore, welfare economics does not seem to offer a clear argument for either approach.
Irrelevant arguments
More relevant under this third-level theme, since they do concern the correct valuation of health states, are the veil of ignorance and the vested interests arguments, both stressing the risk that patients valuing their present health states present partisan valuations in order to serve their own interests. In theory, this could present a real problem. However, in practice, these misgivings seem to be unjustified since empirical research shows that patients often provide higher health state values than the general public, which would rather be to their disadvantage in the context of health improving treatments [2].The conventional approach to valuing health states essentially asks respondents for their ‘off the cuff’ ex ante valuation of the states. The resultant values have sometimes been described as social values but this is a misleading term since respondents are being asked to value the states from the perspective of being in the states. The resultant set of health state values is really an average of the individual valuations of those states.