Patient (2013) 6:235–239 DOI 10.1007/s40271-013-0035-y
LEADING ARTICLE
Carer Preferences in Economic Evaluation and Healthcare Decision Making Hareth Al-Janabi • Nikki McCaffrey Julie Ratcliffe
•
Published online: 5 November 2013 Springer International Publishing Switzerland 2013
Abstract The preferences of informal carers are important to capture for healthcare decision making. This paper discusses how these preferences relate to the economic evaluation of health and care interventions. Three main issues are highlighted. First, there is a need to consider carer impact routinely in economic evaluations. Second, more debate is required around the ethical issues stemming from the inclusion of interdependent preferences in healthcare decision making. Third, there are a number of situations where carer and patient preferences may conflict and practical ways of representing and handling these conflicts would be useful. Key Points for Decision Makers • Carer ‘impact’ should be considered routinely in economic evaluation • Practical methods are needed to jointly consider carer and patient preferences in healthcare decision making • Incorporating carer impact is likely to change the optimal mix of healthcare services
H. Al-Janabi (&) Health Economics Unit, School of Health and Population Sciences, University of Birmingham, Public Health Building, Edgbaston, Birmingham B15 2TT, UK e-mail: [email protected] N. McCaffrey J. Ratcliffe Flinders Health Economics Group, School of Medicine, Flinders University, Bedford Park, Adelaide, SA 5042, Australia
1 Introduction In contrast to formal care provision, an informal carer is a person, such as a family member, friend or neighbour, who gives regular, ongoing assistance to another person, because of illness or disability, without payment for the care given [1]. The future demand for informal care is predicted to increase in the coming decades due an increase in age-related health conditions and disabilities. In addition, government policies are also seeking to promote ageing in place by allowing older people to remain, and be cared for, in the community for as long as possible [2]. Although evidence indicates that the much of the ‘heavyduty’ informal care tends to be provided by older people [1, 3], younger adults (and children) also provide substantial amounts of informal care and face many challenges in managing these caring duties around work, education and relationships. This article examines the role of informal carer preferences in healthcare decision making, focusing on how information on carers’ preferences can be used in economic evaluations. The article outlines how carer preferences can be measured (Sect. 2), the role of carer preferences in economic evaluation (Sect. 3), potential challenges in incorporating carer preference data in a systematic manner in healthcare decisions (Sects. 4 and 5) and areas for further research (Sect. 6).
2 Measuring Carer Preferences The views and values of citizens are increasingly important in delivering public services [4, 5]. In healthcare, a number of industrialised counties have made moves towards a more personalised and ‘consumer-focused’ delivery of
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healthcare; for example, through the introduction of individualised budgets in the UK and consumer-directed aged care in Australia. The ‘baby boomer’ generation of older people is characterised as having a strong preference to exercise greater control over their own lives coupled with an expectation of greater involvement in healthcare decision making. A second factor driving the demand for more information on preferences is the growing use of economic evaluations in funding decisions for healthcare interventions. Economic evaluation is now established as part of the technology appraisal process in a variety of countries. Most forms of economic evaluation require some information on individuals’ preferences, although the methods vary between countries [6]. As many healthcare interventions are likely to affect informal carers, their preferences are important for economic evaluation. Carer preferences, in the context of economic evaluation, refer to the value carers place on a particular (healthcare) intervention. This value may be elicited directly or indirectly by asking carers about the value of quality of life states defined by an instrument which has been designed to measure the impact of the healthcare intervention on the individual’s quality of life. To capture carer preferences for economic evaluation, a choice-based technique is usually required. Different techniques for measuring preferences; for example, standard gambles, time trade-offs, contingent valuation studies and discrete choice experiments (DCEs), have been reviewed extensively elsewhere [7, 8] and a wide range of methods have been used to capture carer preferences. Different techniques may suit different purposes; for example, standard gambles and time trade-offs for valuing quality-of-life states and contingent valuation for directly valuing healthcare interventions. DCEs are flexible enough to be used to assess preferences for both healthcare interventions and quality-of-life states [9]. DCEs can also be used to study the trade-offs that individuals are prepared to make between the key features of a policy or intervention. Potentially, therefore, DCEs offer a particularly attractive option for assessing carers’ preferences as the caring experience is a complex one, with both positive and negative aspects [10, 11]. For example, an intervention may improve the patient’s health (which may improve the carer’s quality of life) at the expense of extra input from the carer (which may reduce the carer’s quality of life).
3 Economic Evaluation and Informal Care In conducting economic evaluations, formal care provision may be adequately captured through the monetary costs associated with this care. However, as previously highlighted, informal care provision is provided without
H. Al-Janabi et al.
payment. Carers are simultaneously both an input into patient care, and a group upon whom outcomes of the care can fall [12]. The method for capturing carer outcomes and their preferences for these outcomes will differ depending on the type of economic evaluation undertaken. A costbenefit analysis, framed in the welfarist tradition, necessitates the measurement and monetary valuation of all benefits emanating from a particular policy change or the introduction of a healthcare intervention. Carers are likely to place some positive value on polices that directly impact on them, such as respite or training programmes, and therefore these values should be considered in a cost-benefit analysis. However, it is important to note that carers are also likely to value interventions that are aimed at the person they are caring for. This is both because the intervention may impact on their care input and because carer is likely to value improvement in the health of their loved ones (and of course in their own health, if relevant). In theory, the monetary values ascribed to a policy by all of those affected (patients, carers and any other individuals) should be aggregated across the relevant population to derive a measure of the total benefits of the intervention. Although a few studies have measured carers’ willingnessto-pay in different contexts [13, 14], these values have rarely been carried through to cost-benefit analyses used to inform healthcare decision making. One exception is a study that examined carers’ willingness-to-pay for technologies to promote independence amongst dementia patients [15]. The authors’ general finding was that the values placed on the technologies by carers were outweighed by the cost of their provision, suggesting that, overall, funding these technologies may not be an efficient use of resources. Cost-utility analysis involves a comparison of the additional costs of an intervention with the value of the health gain generated. Although the largest health gains from interventions will typically accrue to patients themselves, there are many situations where the health of their carers is also likely to be affected [16–18]. In cost-utility analysis, health gains for carers and patients can either be valued directly by the individual affected, or indirectly through valuing changes in health and/or quality of life using an ‘off-the-shelf’ tariff of index values attached to an instrument specifically designed to measure health status and/or quality of life within the framework of economic evaluation. The latter, indirect, route is often utilised by researchers, particularly when conducting economic evaluations alongside clinical trials, as it is more practical but will not reflect carers’ altruistic preferences for health gains for their loved ones [19]. Despite carer health being emphasised in prominent guidelines [20], to date very few applied economic evaluations have considered the impacts of healthcare interventions on carers’ health. Of those
Carer Preferences
studies that have, a number have found that the carer health impact is an important determinant of the cost effectiveness of the intervention [21]. In some cases, an economic evaluation may be more useful to decision makers if the various costs and benefits are presented in a disaggregated way rather than aggregated into a cost-effectiveness, cost-utility or cost-benefit ratio. Such ‘multi-criteria’ or ‘cost-consequence’ analysis is likely to be useful in clinical areas where costs and benefits fall on multiple individuals and institutions [22]. Cost-consequence analysis also allows outcomes for patients and carers to be displayed using different measures. This may be important as decision makers may be interested in impacts on carers that extend beyond health. Recent advances have seen the development of preferencebased measures of carer outcomes such as the Carer Experience Scale and CarerQoL [23, 24]. Cost-consequence analysis provides a framework for these outcomes to be considered alongside patient health outcomes and a diverse range of costs (for example, see [25]).
4 Interdependencies in Preferences It is implicit in their role that carers will care about the wellbeing of the patient. This caring attitude gives rise to a set of preferences that will be interdependent with the preferences of the patient. When an intervention affects the health or wellbeing of the patient, not only will it generate (dis)utility for the patient, but it will likely generate (dis)utility for the carer too. These ‘caring externalities’ [26] have been documented between members of the general population [27] and, given the typically stronger bonds between carer and patient, the degree of interdependency in their preferences is likely to be even higher. There is evidence, for example, that carers are (hypothetically) willing to make substantial sacrifices in terms of their own health and material wellbeing to improve the health of their loved ones [28–30]. Do such interdependencies in preferences pose a problem for healthcare decision making? From a utilitarian perspective, the answer is largely no. Indeed, in order to comprehensively assess the full benefits of a proposed policy it is important to measure the preferences of all those affected (regardless of whether these are dependent on one another). External or spillover ‘benefits’ of policies for carers (and others), stem from the fact that individuals’ lives are interconnected; therefore, negative or positive impacts on one person also impact on others close to them in both a practical and an emotional sense. However, it is important to note that interdependence between carers and patients should be considered when valuing the costs of policy, as well as the benefits stemming from policy. Under
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conditions of interdependence (or altruism), costs impose disutility, not only on the immediate bearer of the costs, but also on their loved ones [31]. However, there is little, if any, research about the measurement of these impacts. Such interdependencies may mean that greater weight is given to the outcomes for patients with carers, relative to those without carers [32]. As a result, incorporating interdependent preferences in economic evaluations may pose ethical challenges. Nevertheless, it is important to note that there are distributional consequences whatever assumption we make about the weight given to carer preferences. The status quo, which largely appears to be a zero weight for carer preferences, has the distributional consequence that greater priority is attached to conditions that are not associated with informal care, than would be expected if carer preferences were taken into account.
5 Conflicts in Preferences Normally carer and patient preferences would be expected to point in the same direction, but sometimes they may conflict. Some events may be positively valued by the patient but negatively valued by the carer (or vice versa). One potential example is in relation to a change in who provides informal care for the patient. In some circumstances, the carer may need to spend so many hours caring that they find themselves feeling resentful or angry towards the patient. In such instances, carers may strongly desire permanent or temporary respite from their caring role in order to reduce the burden of caring. However, the patient may prefer the provider of their care to be their current informal carer (rather than a formal care provider or another relative, for example). A study conducted in the Netherlands to assess the ‘process utility’ associated with informal care found that patients tended to view a change in the person providing care more negatively than the carers on average [32]. The implication of the study is that a sizeable proportion of caring relationships may feature divergent preferences for the carer and patients in terms of the provider of the informal care. Location of care is also important, with preferences for a home death being higher amongst patients than their carers [33]. Although similar reasoning may underpin both patients’ and carers’ preferences, it is possible that their priorities could vary. For example, family carers of terminally ill cancer patients in Taiwan gave higher priority than patients to carer availability and quality of healthcare when stating their preferred place of death [34]. In an economic evaluation, conflicts in preferences would normally be dealt with by summing together the relevant outcomes or preferences. If an intervention (for example respite care to temporarily relieve the carer of
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their duties) made carers happier, but patients less happy, this could be justified, from a theoretical point of view, so long as (i) there was a gain in happiness overall and (ii) this gain could be justified relative to the cost of the intervention. Clearly, however, there are ethical and practical difficulties in delivering interventions that would go against the wishes (or actively harm) one group, and more thought needs to be given to this area. The situation is further complicated by the fact that many patients will have multiple carers [35, 36], often including both formal and informal, and carers may themselves have conflicting views with regards to the patient’s needs [37].
6 Discussion Although there has been an increased emphasis on patient preferences in healthcare, carer preferences continue to have a very minor role in healthcare decisions. As health policies will increasingly impact on carers, greater effort needs to be made to move towards the routine incorporation of carer outcomes and preferences in economic evaluations. Alongside the practical issue of ensuring carer outcomes and preferences are measured, there is a need to address some challenging methodological aspects of incorporating carer preferences in healthcare decision making. One aspect relates to the interdependence between the preferences of carers and patients. It is important to establish whether carers provide their ‘true’ preferences or whether they are acting as a proxy for the patient (in the sense of saying what they think the patient would want). There is evidence, for example, that carers may modify their preferences to reflect the treatment risks and benefits to the patient [38]. Even if the carer is providing their ‘true’ preferences, these preferences are likely to be heavily interdependent with the patient’s (for reasons outlined earlier). There are likely to be distributional consequences from incorporating carer preferences more systematically. More research is needed to understand what these distributional impacts are likely to be. Priority setting occurs at many levels of the healthcare system and while it may seem unethical to give preference for a treatment to an individual because they have a carer (who would also benefit) over an individual who does not, it may also seem unethical to not give any weight to the preferences and outcomes that fall on carers simply because they are not the immediate recipients of the intervention. Another challenge relates to conflicts in preferences between carers and patients. For the technical process of an economic evaluation this may not pose a big problem if conflicting preferences were simply aggregated to come to a summary judgement about the benefits of the
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intervention. However, more research is required as to how to represent, understand and resolve conflicts in preferences in practice, as these conflicts present challenges in delivering interventions. Such work would benefit from the input of economists and, given the ethical, psychological and medical dimensions to the problem, also those with expertise in other areas of social and medical sciences. There are also judgements that need to be made when aggregating carer and patient preferences [39]. For example, it is difficult to know exactly where the line should be drawn as to whom should count as a carer; for example, should the preferences of groups such as close family members who may not act as the person’s main carer but who may offer help and support from time to time also be considered? What about others groups (such as friends and neighbours) who may also be affected by certain public policies? Further research is required to assess whose preferences should ultimately count for economic evaluation, and the development of mechanisms to bridge the gap between theoretical ideals and what might be practical in reality. It is also important to assess to what extent carer preferences should be weighted differently from patient preferences. In conclusion, there is a strong and growing case for including carer preferences in economic evaluation and healthcare decision making. While challenges exist, it is important that these do not deter further research to measure and incorporate carer preferences in economic evaluation. Acknowledgments The authors are grateful to the two anonymous reviewers for their helpful comments on the manuscript. The authors have no conflicts of interest and received no specific funding in relation to this manuscript. HA acknowledges funding from the UK Medical Research Council for an early career fellowship in the economics of health (G1002334). Author contributions HA drafted the manuscript with input from JR and NM. All authors revised the manuscript following peer review. HA is the guarantor for the overall content of the paper.
References 1. Dahlberg L, Demack S, Bambra C. Age and gender of informal carers: a population-based study in the UK. Health Soc Care Community. 2007;15(5):439–45. 2. Pickard L. Informal care for older people provided by their adult children: projections of supply and demand to 2041 in England. University of Kent; 2008. Report No.: PSSRU Discussion Paper 2515. http://www.pssru.ac.uk/pdf/dp2515.pdf. Accessed 23 Sep 2013 3. Tooth L, Russell A, Lucke J, Byrne G, Lee C, Wilson A, et al. Impact of cognitive and physical impairment on carer burden and quality of life. Qual Life Res. 2008;17:267–73.
Carer Preferences 4. Department of Health. Equity and excellence: liberating the NHS. London: The Stationery Office Limited; 2010. https://www.gov. uk/government/uploads/system/uploads/attachment_data/file/213 823/dh_117794.pdf. Accessed 23 Sep 2013 5. Ryan M. Discrete choice experiments in health care. Br Med J. 2004;328:360–1. 6. Claxton K, Walker S, Palmer S, Sculpher M. Appropriate perspectives for healthcare decisions. York: Centre for Health Economics, University of York; 2010. Report No.: CHE Research Paper 54. http://www.york.ac.uk/media/che/documents/papers/researchpapers/ rp54_appropriate_perspectives_for_health_care_decisions.pdf. Accessed 23 Sep 2013 7. Drummond M, Sculpher M, Torrance G, O’Brien B, Stoddart G. Methods for the economic evaluation of health care programmes. 3rd ed. Oxford: Oxford University Press; 2005. 8. Brazier J, Ratcliffe J, Salomon J, Tsuchiya A. Measuring and valuing health benefits for economic evaluation. Oxford: Oxford University Press; 2007. 9. de Bekker-Grob E, Ryan M, Gerard K. Discrete choice experiments in health economics: a review of the literature. Health Econ. 2012;21(2):145–72. 10. Ratcliffe J, Lester L, Couzner L, Crotty M. An assessment of the relationship between informal caring and quality of life in older community-dwelling adults—more positives than negatives? Health Soc Care Community. 2013;21(1):35–46. 11. Al-Janabi H, Coast J, Flynn T. What do people value when they provide unpaid care to an older person? A meta-ethnography with interview follow-up. Soc Sci Med. 2008;67(1):111–21. 12. Wiseman V. Caring: the neglected health outcome? Or input? Health Policy. 1997;39:43–53. 13. Konig M, Wettstein A. Caring for relative with dementia: willingness-to-pay for a reduction in caregiver’s burden. Expert Rev Pharmacoecon Outcomes Res. 2002;2(6):89–101. 14. Van Den Berg B, Brouwer W, Van Exel J, Koopmanschap M. Economic valuation of informal care: the contingent valuation method applied to informal caregiving. Health Econ. 2005;14:169–83. 15. Duff P, Dolphin C. Cost-benefit analysis of assistive technology to support independence for people with dementia—part 2: results from employing the ENABLE cost-benefit model in practice. Technol Disabil. 2007;19:79–90. 16. Wittenberg E, Prosser L. Disutility of illness for caregivers and families: a systematic review of the literature. Pharmacoeconomics. 2013 Jun;31(6):489-500 17. Schulz R, Beach S. Caregiving as a risk factor for mortality. J Am Med Assoc. 1999;282(23):2215–9. 18. Pinquart M, Sorensen S. Differences between caregivers and noncaregivers in psychological health and physical health: a meta-analysis. Psychol Aging. 2003;18(2):250–67. 19. Prosser L, Grosse S, Wittenberg E. Health utility elicitation: is there still a role for direct methods? Pharmacoeconomics. 2012;30(2):83–6. 20. National Institute for Health and Care Excellence. Guide to the methods of technology appraisal. 2013. http://www.nice.org.uk/ media/D45/1E/GuideToMethodsTechnologyAppraisal2013.pdf. Accessed 23 Sep 2013.
239 21. Goodrich K, Kaambwa B, Al-Janabi H. The inclusion of informal care in applied economic evaluation: a review. Value Health. 2012;15(6):975–81. 22. Coast J. Is economic evaluation in touch with society’s health values? Br Med J. 2004;329:1233–6. 23. Al-Janabi H, Flynn T, Coast J. Estimation of a preference based carer experience scale. Med Decis Making. 2011;31(3):458–68. 24. Hoefman R, Van Exel J, Rose J, van de Wetering L, Brouwer W. A discrete choice experiment to obtain tariffs for valuing informal care situations measured with the CarerQol instrument. Med Decis Making. Epub 2013 Jun 14. 25. Gage H, Kaye J, Owen C, Trend P, Wade D. Evaluating rehabilitation using cost-consequences analysis: an example in Parkinson’s disease. Clin Rehabil. 2006;20:232–8. 26. Culyer A. The nature of the commodity ‘health care’ and its efficient allocation. Oxf Econ Pap. 1971;23(2):189–211. 27. Jacobsson F, Carstensen J, Borgquist L. Caring externalities in health economic evaluation: how are they related to severity of illness? Health Policy. 2005;73:172–82. 28. Prosser L, Ray T, O’Brien M, Kleinman K, Santoli J, Lieu T. Preferences and willingness-to-pay for health states prevented by pneumococcal conjugate vaccine. Pediatrics. 2004;113:283–90. 29. Dickie M, Messman M. Parental altruism and the value of avoiding acute illness: are kids worth more than parents? J Environ Econ Manag. 2004;48:1146–74. 30. Basu A, Dale W, Elstein A, Meltzer D. A time tradeoff method for eliciting partner’s quality of life due to patient’s health states in prostate cancer. Med Decis Making. 2010;30:355–65. 31. Bergstrom T. Benefit-cost in a benevolent society. Am Econ Rev. 2006;96(1):339–51. 32. Basu A, Meltzer D. Implications of spillover effects within the family for medical cost-effectiveness analysis. J Health Econ. 2005;24:751–73. 33. Gomes B, Calazani N, Gysels M, Hall S, Higginson I. Heterogeneity and changes in preference for dying at home: a systematic review. BMC Palliat Care 2013;12:7. 34. Tang S, Liu T, Lai M, McCorkle M. Discrepancy in the preferences of place of death between terminally ill cancer patients and their primary family caregivers in Taiwan. Soc Sci Med. 2005;61(7):1560–6. 35. Neubauer S, Holle R, Menn P, Grossfeld-Schmitz M, Graesel E. Measurement of informal care time in a study of patients with dementia. Int Psychogeriatr. 2008;20:1160–76. 36. Tennstedt S, McKinlay J, Sullivan L. Informal care for frail elders: the role of secondary caregivers. Gerontologist. 1989;29 (5):677–83. 37. Usita P, Hall S, Davis J. Role ambiguity in family caregiving. J Appl Gerontol. 2004;23(1):20–39. 38. Karlawish J, Klocinski M, Merz J, Clark C, Asch D. Caregivers’ preferences for the treatment of people with Alzheimer’s disease. Neurology. 2000;55:1008–14. 39. Al-Janabi H, Flynn T, Coast J. QALYs and carers. Pharmacoeconomics. 2011;29(12):1015–23.
LEADING ARTICLE
Carer Preferences in Economic Evaluation and Healthcare Decision Making Hareth Al-Janabi • Nikki McCaffrey Julie Ratcliffe
•
Published online: 5 November 2013 Springer International Publishing Switzerland 2013
Abstract The preferences of informal carers are important to capture for healthcare decision making. This paper discusses how these preferences relate to the economic evaluation of health and care interventions. Three main issues are highlighted. First, there is a need to consider carer impact routinely in economic evaluations. Second, more debate is required around the ethical issues stemming from the inclusion of interdependent preferences in healthcare decision making. Third, there are a number of situations where carer and patient preferences may conflict and practical ways of representing and handling these conflicts would be useful. Key Points for Decision Makers • Carer ‘impact’ should be considered routinely in economic evaluation • Practical methods are needed to jointly consider carer and patient preferences in healthcare decision making • Incorporating carer impact is likely to change the optimal mix of healthcare services
H. Al-Janabi (&) Health Economics Unit, School of Health and Population Sciences, University of Birmingham, Public Health Building, Edgbaston, Birmingham B15 2TT, UK e-mail: [email protected] N. McCaffrey J. Ratcliffe Flinders Health Economics Group, School of Medicine, Flinders University, Bedford Park, Adelaide, SA 5042, Australia
1 Introduction In contrast to formal care provision, an informal carer is a person, such as a family member, friend or neighbour, who gives regular, ongoing assistance to another person, because of illness or disability, without payment for the care given [1]. The future demand for informal care is predicted to increase in the coming decades due an increase in age-related health conditions and disabilities. In addition, government policies are also seeking to promote ageing in place by allowing older people to remain, and be cared for, in the community for as long as possible [2]. Although evidence indicates that the much of the ‘heavyduty’ informal care tends to be provided by older people [1, 3], younger adults (and children) also provide substantial amounts of informal care and face many challenges in managing these caring duties around work, education and relationships. This article examines the role of informal carer preferences in healthcare decision making, focusing on how information on carers’ preferences can be used in economic evaluations. The article outlines how carer preferences can be measured (Sect. 2), the role of carer preferences in economic evaluation (Sect. 3), potential challenges in incorporating carer preference data in a systematic manner in healthcare decisions (Sects. 4 and 5) and areas for further research (Sect. 6).
2 Measuring Carer Preferences The views and values of citizens are increasingly important in delivering public services [4, 5]. In healthcare, a number of industrialised counties have made moves towards a more personalised and ‘consumer-focused’ delivery of
236
healthcare; for example, through the introduction of individualised budgets in the UK and consumer-directed aged care in Australia. The ‘baby boomer’ generation of older people is characterised as having a strong preference to exercise greater control over their own lives coupled with an expectation of greater involvement in healthcare decision making. A second factor driving the demand for more information on preferences is the growing use of economic evaluations in funding decisions for healthcare interventions. Economic evaluation is now established as part of the technology appraisal process in a variety of countries. Most forms of economic evaluation require some information on individuals’ preferences, although the methods vary between countries [6]. As many healthcare interventions are likely to affect informal carers, their preferences are important for economic evaluation. Carer preferences, in the context of economic evaluation, refer to the value carers place on a particular (healthcare) intervention. This value may be elicited directly or indirectly by asking carers about the value of quality of life states defined by an instrument which has been designed to measure the impact of the healthcare intervention on the individual’s quality of life. To capture carer preferences for economic evaluation, a choice-based technique is usually required. Different techniques for measuring preferences; for example, standard gambles, time trade-offs, contingent valuation studies and discrete choice experiments (DCEs), have been reviewed extensively elsewhere [7, 8] and a wide range of methods have been used to capture carer preferences. Different techniques may suit different purposes; for example, standard gambles and time trade-offs for valuing quality-of-life states and contingent valuation for directly valuing healthcare interventions. DCEs are flexible enough to be used to assess preferences for both healthcare interventions and quality-of-life states [9]. DCEs can also be used to study the trade-offs that individuals are prepared to make between the key features of a policy or intervention. Potentially, therefore, DCEs offer a particularly attractive option for assessing carers’ preferences as the caring experience is a complex one, with both positive and negative aspects [10, 11]. For example, an intervention may improve the patient’s health (which may improve the carer’s quality of life) at the expense of extra input from the carer (which may reduce the carer’s quality of life).
3 Economic Evaluation and Informal Care In conducting economic evaluations, formal care provision may be adequately captured through the monetary costs associated with this care. However, as previously highlighted, informal care provision is provided without
H. Al-Janabi et al.
payment. Carers are simultaneously both an input into patient care, and a group upon whom outcomes of the care can fall [12]. The method for capturing carer outcomes and their preferences for these outcomes will differ depending on the type of economic evaluation undertaken. A costbenefit analysis, framed in the welfarist tradition, necessitates the measurement and monetary valuation of all benefits emanating from a particular policy change or the introduction of a healthcare intervention. Carers are likely to place some positive value on polices that directly impact on them, such as respite or training programmes, and therefore these values should be considered in a cost-benefit analysis. However, it is important to note that carers are also likely to value interventions that are aimed at the person they are caring for. This is both because the intervention may impact on their care input and because carer is likely to value improvement in the health of their loved ones (and of course in their own health, if relevant). In theory, the monetary values ascribed to a policy by all of those affected (patients, carers and any other individuals) should be aggregated across the relevant population to derive a measure of the total benefits of the intervention. Although a few studies have measured carers’ willingnessto-pay in different contexts [13, 14], these values have rarely been carried through to cost-benefit analyses used to inform healthcare decision making. One exception is a study that examined carers’ willingness-to-pay for technologies to promote independence amongst dementia patients [15]. The authors’ general finding was that the values placed on the technologies by carers were outweighed by the cost of their provision, suggesting that, overall, funding these technologies may not be an efficient use of resources. Cost-utility analysis involves a comparison of the additional costs of an intervention with the value of the health gain generated. Although the largest health gains from interventions will typically accrue to patients themselves, there are many situations where the health of their carers is also likely to be affected [16–18]. In cost-utility analysis, health gains for carers and patients can either be valued directly by the individual affected, or indirectly through valuing changes in health and/or quality of life using an ‘off-the-shelf’ tariff of index values attached to an instrument specifically designed to measure health status and/or quality of life within the framework of economic evaluation. The latter, indirect, route is often utilised by researchers, particularly when conducting economic evaluations alongside clinical trials, as it is more practical but will not reflect carers’ altruistic preferences for health gains for their loved ones [19]. Despite carer health being emphasised in prominent guidelines [20], to date very few applied economic evaluations have considered the impacts of healthcare interventions on carers’ health. Of those
Carer Preferences
studies that have, a number have found that the carer health impact is an important determinant of the cost effectiveness of the intervention [21]. In some cases, an economic evaluation may be more useful to decision makers if the various costs and benefits are presented in a disaggregated way rather than aggregated into a cost-effectiveness, cost-utility or cost-benefit ratio. Such ‘multi-criteria’ or ‘cost-consequence’ analysis is likely to be useful in clinical areas where costs and benefits fall on multiple individuals and institutions [22]. Cost-consequence analysis also allows outcomes for patients and carers to be displayed using different measures. This may be important as decision makers may be interested in impacts on carers that extend beyond health. Recent advances have seen the development of preferencebased measures of carer outcomes such as the Carer Experience Scale and CarerQoL [23, 24]. Cost-consequence analysis provides a framework for these outcomes to be considered alongside patient health outcomes and a diverse range of costs (for example, see [25]).
4 Interdependencies in Preferences It is implicit in their role that carers will care about the wellbeing of the patient. This caring attitude gives rise to a set of preferences that will be interdependent with the preferences of the patient. When an intervention affects the health or wellbeing of the patient, not only will it generate (dis)utility for the patient, but it will likely generate (dis)utility for the carer too. These ‘caring externalities’ [26] have been documented between members of the general population [27] and, given the typically stronger bonds between carer and patient, the degree of interdependency in their preferences is likely to be even higher. There is evidence, for example, that carers are (hypothetically) willing to make substantial sacrifices in terms of their own health and material wellbeing to improve the health of their loved ones [28–30]. Do such interdependencies in preferences pose a problem for healthcare decision making? From a utilitarian perspective, the answer is largely no. Indeed, in order to comprehensively assess the full benefits of a proposed policy it is important to measure the preferences of all those affected (regardless of whether these are dependent on one another). External or spillover ‘benefits’ of policies for carers (and others), stem from the fact that individuals’ lives are interconnected; therefore, negative or positive impacts on one person also impact on others close to them in both a practical and an emotional sense. However, it is important to note that interdependence between carers and patients should be considered when valuing the costs of policy, as well as the benefits stemming from policy. Under
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conditions of interdependence (or altruism), costs impose disutility, not only on the immediate bearer of the costs, but also on their loved ones [31]. However, there is little, if any, research about the measurement of these impacts. Such interdependencies may mean that greater weight is given to the outcomes for patients with carers, relative to those without carers [32]. As a result, incorporating interdependent preferences in economic evaluations may pose ethical challenges. Nevertheless, it is important to note that there are distributional consequences whatever assumption we make about the weight given to carer preferences. The status quo, which largely appears to be a zero weight for carer preferences, has the distributional consequence that greater priority is attached to conditions that are not associated with informal care, than would be expected if carer preferences were taken into account.
5 Conflicts in Preferences Normally carer and patient preferences would be expected to point in the same direction, but sometimes they may conflict. Some events may be positively valued by the patient but negatively valued by the carer (or vice versa). One potential example is in relation to a change in who provides informal care for the patient. In some circumstances, the carer may need to spend so many hours caring that they find themselves feeling resentful or angry towards the patient. In such instances, carers may strongly desire permanent or temporary respite from their caring role in order to reduce the burden of caring. However, the patient may prefer the provider of their care to be their current informal carer (rather than a formal care provider or another relative, for example). A study conducted in the Netherlands to assess the ‘process utility’ associated with informal care found that patients tended to view a change in the person providing care more negatively than the carers on average [32]. The implication of the study is that a sizeable proportion of caring relationships may feature divergent preferences for the carer and patients in terms of the provider of the informal care. Location of care is also important, with preferences for a home death being higher amongst patients than their carers [33]. Although similar reasoning may underpin both patients’ and carers’ preferences, it is possible that their priorities could vary. For example, family carers of terminally ill cancer patients in Taiwan gave higher priority than patients to carer availability and quality of healthcare when stating their preferred place of death [34]. In an economic evaluation, conflicts in preferences would normally be dealt with by summing together the relevant outcomes or preferences. If an intervention (for example respite care to temporarily relieve the carer of
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their duties) made carers happier, but patients less happy, this could be justified, from a theoretical point of view, so long as (i) there was a gain in happiness overall and (ii) this gain could be justified relative to the cost of the intervention. Clearly, however, there are ethical and practical difficulties in delivering interventions that would go against the wishes (or actively harm) one group, and more thought needs to be given to this area. The situation is further complicated by the fact that many patients will have multiple carers [35, 36], often including both formal and informal, and carers may themselves have conflicting views with regards to the patient’s needs [37].
6 Discussion Although there has been an increased emphasis on patient preferences in healthcare, carer preferences continue to have a very minor role in healthcare decisions. As health policies will increasingly impact on carers, greater effort needs to be made to move towards the routine incorporation of carer outcomes and preferences in economic evaluations. Alongside the practical issue of ensuring carer outcomes and preferences are measured, there is a need to address some challenging methodological aspects of incorporating carer preferences in healthcare decision making. One aspect relates to the interdependence between the preferences of carers and patients. It is important to establish whether carers provide their ‘true’ preferences or whether they are acting as a proxy for the patient (in the sense of saying what they think the patient would want). There is evidence, for example, that carers may modify their preferences to reflect the treatment risks and benefits to the patient [38]. Even if the carer is providing their ‘true’ preferences, these preferences are likely to be heavily interdependent with the patient’s (for reasons outlined earlier). There are likely to be distributional consequences from incorporating carer preferences more systematically. More research is needed to understand what these distributional impacts are likely to be. Priority setting occurs at many levels of the healthcare system and while it may seem unethical to give preference for a treatment to an individual because they have a carer (who would also benefit) over an individual who does not, it may also seem unethical to not give any weight to the preferences and outcomes that fall on carers simply because they are not the immediate recipients of the intervention. Another challenge relates to conflicts in preferences between carers and patients. For the technical process of an economic evaluation this may not pose a big problem if conflicting preferences were simply aggregated to come to a summary judgement about the benefits of the
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intervention. However, more research is required as to how to represent, understand and resolve conflicts in preferences in practice, as these conflicts present challenges in delivering interventions. Such work would benefit from the input of economists and, given the ethical, psychological and medical dimensions to the problem, also those with expertise in other areas of social and medical sciences. There are also judgements that need to be made when aggregating carer and patient preferences [39]. For example, it is difficult to know exactly where the line should be drawn as to whom should count as a carer; for example, should the preferences of groups such as close family members who may not act as the person’s main carer but who may offer help and support from time to time also be considered? What about others groups (such as friends and neighbours) who may also be affected by certain public policies? Further research is required to assess whose preferences should ultimately count for economic evaluation, and the development of mechanisms to bridge the gap between theoretical ideals and what might be practical in reality. It is also important to assess to what extent carer preferences should be weighted differently from patient preferences. In conclusion, there is a strong and growing case for including carer preferences in economic evaluation and healthcare decision making. While challenges exist, it is important that these do not deter further research to measure and incorporate carer preferences in economic evaluation. Acknowledgments The authors are grateful to the two anonymous reviewers for their helpful comments on the manuscript. The authors have no conflicts of interest and received no specific funding in relation to this manuscript. HA acknowledges funding from the UK Medical Research Council for an early career fellowship in the economics of health (G1002334). Author contributions HA drafted the manuscript with input from JR and NM. All authors revised the manuscript following peer review. HA is the guarantor for the overall content of the paper.
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