The Feasibility and Validity of Preference-Based Quality of Life Measures With Informal Carers: A Think-Aloud Study.

OBJECTIVES: A range of preference-based quality of life (QoL) measures have been proposed for use with informal carers. Qualitative evaluation of validity and feasibility of the measures is an important step in understanding whether measures will work as intended. At present, little is known about the performance of different types of preference-based QoL measures with informal carers. The objective of this study was to qualitatively assess the feasibility, content validity (including face validity), and acceptability of 5 QoL measures (the Carer Experience Scale, CarerQoL-7D, ASCOT-C, ICECAP-A, and EQ-5D-5L) with informal carers. METHODS: A total of 24 "think-aloud" interviews were conducted with a cross-section of carers of adults in the United Kingdom. This think-aloud process was followed by semistructured discussion to probe issues of validity and feasibility in more detail. The interview data were transcribed, coded to identify the frequency of errors in completing the QoL measures and thematically analyzed to study the validity, feasibility, and acceptability of the measures. RESULTS: Few errors (3%-7% per item) were identified in completing each of the measures with little distinct pattern. Most participants found the measures to be concise, clear, and relevant. Challenges included relevance, context, time period, missing items, multiple questions, and response options. Informal carers generally expressed a preference for using a care-related QoL measure. CONCLUSIONS: Existing preference-based QoL measures have encouraging validity and feasibility within a mixed sample of informal carers, with minor challenges raised. These challenges ought to be considered, alongside the decision context, when administering QoL measures in this context.

The use of composite time trade-off and discrete choice experiment methods for the valuation of the Short Warwick-Edinburgh Mental Well-being Scale (SWEMWBS): a think-aloud study.

PURPOSE: To identify patterns and problems in completing composite time trade-off (C-TTO) and discrete choice experiment (DCE) exercises for the valuation of the Short Warwick-Edinburgh Mental Well-being Scale (SWEMWBS) to inform the optimisation of a valuation protocol. METHODS: Fourteen cognitive interviews were conducted in the UK using concurrent and retrospective think-aloud and probing techniques. Each participant completed 8 C-TTO tasks and 8 DCE tasks within a computer-assisted personal interview setting. Verbal information was transcribed verbatim. Axial coding and thematic analysis were used to organise the qualitative data and identify patterns and problems with the completion of tasks. RESULTS: While participants found the tasks generally manageable, five broad themes emerged to explain and optimise the response to the tasks. (1) Format and structure: attention to the design of practice examples, instructions, and layout were needed. (2) Items and levels: underlying relationships were discovered across different combinations of levels of SWEMWBS items. (3) Decision heuristics: participants engaged in diverse strategies to assist trade-off decisions. (4) Valuation feasibility: certain states were difficult to imagine, compare and quantify. (5) Valuation outcome: the data quality was affected by participants' discriminatory ability across states and their time trade-off decisions. CONCLUSION: The interviews contributed insights regarding the robustness of the proposed methods. The application of C-TTO and DCE valuation techniques was practical and suitable for capturing individual attitudes towards different mental well-being scenarios. A modified protocol informed by the results is being tested in a larger sample across the UK.

Estimating an exchange-rate between care-related and health-related quality of life outcomes for economic evaluation: An application of the wellbeing valuation method.

Quality of life outcomes for family carers and patients may be measured in different ways within the same economic evaluation. We used the wellbeing valuation method to calculate "exchange rates" between care-related outcomes (the Carer Experience Scale and CarerQoL-7D) and health-related (the EQ-5D-5L) outcomes. Data on quality of life outcomes were collected through a postal quality of life survey in the UK. A random effects model was used to estimate carers' wellbeing as a function of their EQ-5D-5L, Carer Experience Scale (or CarerQoL-7D) and a set of control variables. When life satisfaction was used as the measure of wellbeing, a one-point gain in the Carer Experience Scale (0-100 scale) was equivalent (in wellbeing terms) to a 0.014 gain in EQ-5D-5L value; and a one point gain in the CarerQoL-7D (0-100 scale) was equivalent to a 0.033 gain in EQ-5D-5L. The exchange rate values were reduced when capability was used as the measure of wellbeing. The exchange rates estimated in this study offer a means to place carer and patient outcomes, measured via different quality of life instruments, on a common scale, although there are important issues to consider in operationalising the technique.

The scope of carer effects and their inclusion in decision-making: a UK-based Delphi study.

BACKGROUND AND OBJECTIVE: Health and social care may affect unpaid (family) carers' health and wellbeing in addition to patients' lives. It is recommended that such impacts (carer effects) are considered in decision-making. However, the scope of carer effects and range of decisions where carer effects should be considered is uncertain. This study aimed to identify: (i) how different categories of healthcare and social care were perceived to impact on unpaid carers; and (ii) whether there was consensus about when carer effects should be formally considered in decision-making contexts. METHODS: A two round, online Delphi study was conducted with 65 UK-based participants (unpaid carers, care professionals, and researchers) with expertise in dementia, mental health, and stroke. Participants considered two broad forms of 'interventions' (patient treatment and replacement care) and two broad forms of 'organisational change' (staffing and changes in timing/location of care). Participants assessed the likely impacts of these on unpaid carers and whether impacts should be considered in decision-making. RESULTS: Participants predicted interventions and organisational changes would impact on multiple domains of unpaid carers' lives, with 'emotional health' the most likely outcome to be affected. Patient treatment and replacement care services ('interventions') were associated with positive impacts across all domains. Conversely, timing/location changes and staffing changes ('organisational changes') were perceived to have mixed and negative impacts. There was widespread support (80-81 %) for considering carer effects in research studies, funding decisions, and patient decision-making. CONCLUSIONS: This study highlights a perception that carer effects are widespread and important to consider in economic evaluation and decision-making. It highlights the particular need to measure and value effects on carers' emotional health and the need to use a societal perspective to avoid cost shifting to unpaid carers when introducing interventions and making organisational changes.

Validity and Responsiveness of Preference-Based Quality-of-Life Measures in Informal Carers: A Comparison of 5 Measures Across 4 Conditions.

OBJECTIVES: Carer quality-of-life (QoL) effects are recommended for inclusion in economic evaluations, but little is known about the relative performance of different types of QoL measures with carers. This study evaluated the validity and responsiveness of 3 care-related QoL measures (the Carer Experience Scale [CES], CarerQoL-7D, and ASCOT-Carer), 1 health-related QoL measure (the EQ-5D-5L), and 1 generic QoL measure (the ICECAP-A). METHODS: Validity and responsiveness were assessed in a UK sample of informal carers of adults with dementia, stroke, mental illness, or rheumatoid arthritis. A questionnaire containing the 5 QoL measures was posted to carers identified through the Family Resources Survey (N = 1004). Hypotheses regarding the anticipated associations between constructs related to the QoL of carers were tested to investigate construct validity and responsiveness. RESULTS: Each measure exhibited some level of construct validity. In general, larger effect sizes and stronger associations were detected for the ASCOT-Carer and ICECAP-A measures in the pooled sample and across all conditions. The 5 measures did not exhibit clear responsiveness to changes over a 12-month period in care recipient health status or hours of care provided per week. CONCLUSION: The results of this study provide initial evidence of the validity of care-related, health-related, and generic QoL (capability) measures in informal carers of adults with 4 highly prevalent conditions. Care-related measures were not always more sensitive to constructs associated with QoL of carers compared with generic measures. The performance of the ICECAP-A was comparable with that of the best-performing care-related measure, the ASCOT-Carer.

UK General Population Utility Values for the SIDECAR-D Instrument Measuring the Impact of Caring for People With Dementia.

OBJECTIVES: Dementia affects many people, with numbers expected to grow as populations age. Many people with dementia receive informal/family/unpaid care, for example, from a spouse or child, which may affect carer quality of life. Measuring the effectiveness of health/social care interventions for carers requires a value measure of the quality-of-life impact of caring. This motivated development of the Scales Measuring the Impact of Dementia on Carers-D (SIDECAR-D) instrument. This study aimed to obtain general population values for SIDECAR-D to aid incorporating the impact of caring in economic evaluation. METHODS: Members of the UK general public completed a best-worst scaling object case survey, which included the 18 SIDECAR-D items and EQ-5D-3L descriptions. Responses were analyzed using scale-adjusted finite mixture models. Relative importance scores (RISs) for the 18 SIDECAR-D items formed the SIDECAR-D relative scale measuring the relative impact of caring. The SIDECAR-D tariff, on the full health = 1, dead = 0 scale, was derived by rescaling EQ-5D-3L and SIDECAR-D RISs so the EQ-5D-3L RISs equaled anchored valuations of the EQ-5D-3L pits state from a visual analog scale task. RESULTS: Five hundred ten respondents completed the survey. The model had 2 parameter and 3 scale classes. Additive utility decrements of SIDECAR-D items ranged from -0.05 to -0.162. Utility scores range from 0.95 for someone affirming 1 item to -0.297 for someone affirming all 18. CONCLUSION: SIDECAR-D is a needs-based scale of the impact on quality of life of caring for someone with dementia, with a valuation tariff to support its use in economic evaluation.

Parental health spillover effects of paediatric rare genetic conditions.

PURPOSE: The complexity and severity of rare genetic conditions pose substantial burden to families. While the importance of spillovers on carers' health in resource allocation decisions is increasingly recognised, there is significant lack of empirical evidence in the context of rare diseases. The objective of this study was to estimate the health spillovers of paediatric rare genetic conditions on parents. METHODS: Health-related quality-of-life (HRQoL) data from children with rare genetic conditions (genetic kidney diseases, mitochondrial diseases, epileptic encephalopathies, brain malformations) and their parents were collected using the CHU9D and SF-12 measures, respectively. We used two approaches to estimate parental health spillovers. To quantify the 'absolute health spillover', we matched our parent cohort to the Australian general population. To quantify the 'relative health spillover', regression models were applied using the cohort data. RESULTS: Parents of affected children had significantly lower HRQoL compared to matched parents in the general public (- 0.06; 95% CIs - 0.08, - 0.04). Multivariable regression demonstrated a positive association between parental and child health. The mean magnitude of HRQoL loss in parents was estimated to be 33% of the HRQoL loss observed in children (95% CIs 21%, 46%). CONCLUSION: Paediatric rare genetic conditions appear to be associated with substantial parental health spillovers. This highlights the importance of including health effects on family members and caregivers into economic evaluation of genomic technologies and personalised medicine. Overlooking spillover effects may undervalue the benefits of diagnosis and management in this context. This study also expands the knowledge of family spillover to the rare disease spectrum.

Parents' preferences for nursery care when children are unwell: a discrete choice experiment.

BACKGROUND: Pre-school children's daycare is associated with increased incidence of respiratory and diarrhoeal illnesses. While the incidence might be reduced if all unwell children were kept at home, parental employment pressures make this difficult when children are marginally unwell. METHODS: A discrete choice experiment (DCE) was conducted to identify what aspects of daycare policy and provision would affect parents' decisions to keep marginally unwell children home. Prior qualitative research informed parameter choice. The DCE was accompanied by a best-worst scaling task examining preferences for four modifiable aspects of care: swapping unused daycare sessions, reimbursing unused sessions, daycare paracetamol policy and presence of a 'quiet room'. RESULTS: Paracetamol guidelines and the presence of a quiet room had the strongest predicted influence on parents' decision-making. Conditional on assumptions about the set-up of the daycare, introducing a 'no paracetamol' policy would result in a fall from 62 to 25% in mean predicted probabilities of a parent sending a marginally unwell child to nursery, while introducing a quiet room would increase the mean probability from 34 to 53%. CONCLUSIONS: Daycare policy, particularly the use of paracetamol prior to attendance, could impact parents' decisions to send unwell children to daycare, potentially influencing the transmission of children's infectious illness.

Incorporating Household Spillovers in Cost Utility Analysis: A Case Study Using Behavior Change in COPD.

OBJECTIVES: It is important to capture all health effects of interventions in cost-utility analyses conducted under a societal or healthcare perspective. However, this is rarely done. Household spillovers (health effects on patients' other household members) may be particularly likely in the context of technologies and interventions to change behaviors that are interdependent in the household. Our objective was to prospectively collect outcome data from household members and illustrate how these can be included in a cost-utility analysis of a behavior change intervention in chronic obstructive pulmonary disease (COPD). METHODS: Data were collected from patients' household members (n = 153) alongside a randomized controlled trial of a COPD self-management intervention. The impact of the intervention on household members' EQ-5D-5L scores (primary outcome), was evaluated. Analyses were then carried out to estimate household members' quality-adjusted life-years (QALYs) and assess the impact of including these QALYs on cost-effectiveness. RESULTS: The intervention had a negligible spillover on household members' EQ-5D-5L scores (-0.007; p = .75). There were also no statistically significant spillovers at the 5 percent level in household member secondary outcomes. In the base-case model, the inclusion of household member QALYs in the incremental cost-effectiveness ratio (ICER) denominator marginally increased the ICER from GBP 10,271 (EUR 13,146) to GBP 10,991 (EUR 14,068) per QALY gained. CONCLUSIONS: This study demonstrates it is feasible to prospectively collect and include household members' outcome data in cost utility analysis, although the study highlighted several methodological issues. In this case, the intervention did not impact significantly on household members' health or health behaviors, but inclusion of household spillovers may make a difference in other contexts.

Do capability and functioning differ? A study of U.K. survey responses.

A core feature of the capability approach is that a person's capabilities (what they are able to do and be in their life) can differ from their functionings (what they actually do and are in their life). However, the degree to which capability and functioning differ in practice is unclear. This paper investigates this issue, focusing on capability and functioning differences (CFD) across different aspects of life and different individuals. In the study, the ICECAP-A capability questionnaire was modified to measure both functionings and capabilities and was completed by U.K.-based convenience sample of 943 people. Around one third of people reported CFD in at least one area of their life, most commonly in terms of their "achievement." People were more likely to report CFD when they had a degree-level education and when they had impaired health. An additional finding was that capability varied more with education whereas functioning varied more with health status. This finding needs further examination, but it suggests that the choice of evaluative space may influence how priorities are set for public spending.

Informal care: choice or constraint?

BACKGROUND: 'Choice' is increasingly pursued as a goal of social policy. However, the degree to which choice is exercised when entering an informal caring role is open to debate. AIM: In this study, we examined the degree of choice and constraint in entering a caring role, and the relationship between choice and carers' well-being. METHODS: Data were derived from 1100 responses to a postal survey conducted in a British city. Statistical tests of association and multivariable regression modelling were applied to study the factors associated with choice in entering a caring role and the association that choice in entering a caring role had with carers' well-being. RESULTS: We found that informal care was generally perceived to be a free choice, albeit in most cases, a choice was also constrained by duty, financial or social resources. Having a sense of free choice in entering care was strongly and positively associated with the carer's well-being. CONCLUSION: The study findings are consistent with a view that enabling individuals to have more choice in their caring roles may be beneficial.

Assessing the validity of the ICECAP-A capability measure for adults with depression.

BACKGROUND: Effectiveness and cost-effectiveness are increasingly important considerations in determining which mental health services are funded. Questions have been raised concerning the validity of generic health status instruments used in economic evaluation for assessing mental health problems such as depression; measuring capability wellbeing offers a possible alternative. The aim of this study is to assess the validity of the ICECAP-A capability instrument for individuals with depression. METHODS: Hypotheses were developed using concept mapping. Validity tests and multivariable regression analysis were applied to data from a cross-sectional dataset to assess the performance of ICECAP-A in individuals who reported having a primary condition of depression. The ICECAP-A was collected alongside instruments used to measure: 1. depression using the depression scale of the Depression, Anxiety and Stress Scale (DASS-D of DASS-21); 2. mental health using the Kessler Psychological Distress Scale (K10); 3. generic health status using a common measure collected for use in economic evaluations, the five level version of EQ-5D (EQ-5D-5L). RESULTS: Hypothesised associations between the ICECAP-A (items and index scores) and depression constructs were fully supported in statistical tests. In the multivariable analysis, instruments designed specifically to measure depression and mental health explained a greater proportion of the variation in ICECAP-A than the EQ-5D-5L. CONCLUSION: The ICECAP-A instrument appears to be suitable for assessing outcome in adults with depression for resource allocation purposes. Further research is required on its responsiveness and use in economic evaluation. Using a capability perspective when assessing cost-effectiveness could potentially re-orientate resource provision across physical and mental health care services.

Development of a measure (ICECAP-Close Person Measure) through qualitative methods to capture the benefits of end-of-life care to those close to the dying for use in economic evaluation.

BACKGROUND: End-of-life care affects both the patient and those close to them. Typically, those close to the patient are not considered within economic evaluation, which may lead to the omission of important benefits resulting from end-of-life care. AIM: To develop an outcome measure suitable for use in economic evaluation that captures the benefits of end-of-life care to those close to the dying. DESIGN: To develop the descriptive system for the outcome measure, in-depth qualitative interviews were conducted with the participants and constant comparative analysis methods were used to develop a descriptive system for the measure. PARTICIPANTS: Twenty-seven individuals bereaved within the last 2 years or with a close-person currently receiving end-of-life care were purposively recruited into the study. Participants were recruited through newsletters, adverts, snowball sampling and a local hospice. RESULTS: Twenty-seven individuals were recruited. A measure of capability with six attributes, each with five levels, was developed based on themes arising from the analysis. Attributes comprise the following: good communication with services, privacy and space to be with the loved one, emotional support, practical support, being able to prepare and cope and being free from emotional distress related to the condition of the decedent. CONCLUSION: This measure is designed to capture the benefits of end-of-life care to close-persons for use in economic evaluation. Further research should value the measure and develop methods for incorporating outcomes for close-persons into economic evaluation.

Measuring Health Spillovers for Economic Evaluation: A Case Study in Meningitis.

The health of carers and others close to the patient will often be relevant to economic evaluation, but it is very rarely considered in practice. This may reflect a lack of understanding of how the spillover effect of illness can be appropriately quantified. In this study we used three different approaches to quantify health spillovers resulting from meningitis. We conducted a survey of 1218 family networks affected by meningitis and used regression modelling to estimate spillover effects. The findings show that meningitis had long-term effects on family members' health, particularly affecting the likelihood of family members reporting anxiety and depression. These effects extended beyond a single close family member. These findings suggest that vaccinating against meningitis will bring significant health benefits not just to those that might have contracted the illness but also to their family networks. In methodological terms, different approaches for quantifying health spillovers provided broadly consistent results. The choice of method will be influenced by the ease of collecting primary data from family members in intervention contexts. © 2015 The Authors. Health Economics published by John Wiley & Sons Ltd.

The validity and responsiveness of the ICECAP-A capability-well-being measure in women with irritative lower urinary tract symptoms.

PURPOSE: A desire to incorporate broader aspects of well-being in health economic evaluations has led to the development of the ICEpop CAPability measure for Adults (ICECAP-A). The ICECAP-A draws upon Amartya Sen's capability approach and conceptualises well-being as the capability to achieve Stability, Attachment, Autonomy, Achievement, and Enjoyment. The aim of this study was to assess the psychometric performance of the ICECAP-A in a context where patient outcomes can extend beyond health-related quality of life. METHODS: Longitudinal data were collected for 478 women with symptoms of urinary frequency and urgency, with or without incontinence. Women were recruited across 22 hospitals in the UK and had a mean age of 55 (SD 14). The psychometric performance of the measure was evaluated in relation to the EuroQol Five-Dimension Questionnaire (EQ-5D-3L) and the International Consultation on Incontinence Questionnaire for Overactive Bladder (ICIQ-OAB) and involved an assessment of acceptability, construct validity, and responsiveness using parametric and nonparametric methods. RESULTS: ICECAP-A showed good convergence with the ICIQ-OAB with 20 out of 22 expected patterns of relationship confirmed. Findings suggested that the ICECAP-A has better discriminative properties than EQ-5D-3L and as good as those of the ICIQ-OAB, confirming expected associations with clinical and demographic factors. The ICECAP-A was more responsive than EQ-5D-3L and ICIQ-OAB to deteriorations of clinical symptoms. Improvements in symptoms were not valued as highly as deteriorations by either ICECAP-A or EQ-5D-3L. CONCLUSIONS: The ICECAP-A is a valid and responsive measure capturing broad emotional and practical impacts of urinary symptoms on women's well-being and could be considered for use in economic evaluations in this context.

Factors influencing parents' decision-making when sending children with respiratory tract infections to nursery.

BACKGROUND: Many families rely on formal day care provision, which can be problematic when children are unwell. Attendance in these circumstances may impact on the transmission of infections in both day care and the wider community. METHODS: Thirty-one semi-structured interviews were conducted to investigate how parents make decisions about nursery care when children are unwell. Topics for discussion included: illness attitudes, current practice during childhood illness and potential nursery policy changes that could affect decision-making. RESULTS: A combination of illness perceptions and external factors affected decision-making. Parents: (i) considered the severity of respiratory and non-respiratory symptoms differently, and stated that while most other contagious illnesses required nursery exclusion, coughs/colds did not; (ii) said decisions were not solely based on nursery policy, but on practical challenges such as work absences, financial penalties and alternative care availability; (iii) identified modifiable nursery policy factors that could potentially help parents keep unwell children at home, potentially reducing transmission of infectious illness. CONCLUSIONS: Decision-making is a complex interaction between the child's illness, personal circumstance and nursery policy. Improving our understanding of the modifiable aspects of nursery policies and the extent to which these factors affect decision-making could inform the design and implementation of interventions to reduce the transmission of infectious illness and the associated burden on NHS services.

Test-retest reliability of capability measurement in the UK general population.

Although philosophically attractive, it may be difficult, in practice, to measure individuals' capabilities (what they are able to do in their lives) as opposed to their functionings (what they actually do). To examine whether capability information could be reliably self-reported, we administered a measure of self-reported capability (the Investigating Choice Experiments Capability Measure for Adults, ICECAP-A) on two occasions, 2 weeks apart, alongside a self-reported health measure (the EuroQol Five Dimensional Questionnaire with 3 levels, EQ-5D-3L). We found that respondents were able to report capabilities with a moderate level of consistency, although somewhat less reliably than their health status. The more socially orientated nature of some of the capability questions may account for the difference.

Scoring the Icecap-a capability instrument. Estimation of a UK general population tariff.

This paper reports the results of a best-worst scaling (BWS) study to value the Investigating Choice Experiments Capability Measure for Adults (ICECAP-A), a new capability measure among adults, in a UK setting. A main effects plan plus its foldover was used to estimate weights for each of the four levels of all five attributes. The BWS study was administered to 413 randomly sampled individuals, together with sociodemographic and other questions. Scale-adjusted latent class analyses identified two preference and two (variance) scale classes. Ability to characterize preference and scale heterogeneity was limited, but data quality was good, and the final model exhibited a high pseudo-r-squared. After adjusting for heterogeneity, a population tariff was estimated. This showed that 'attachment' and 'stability' each account for around 22% of the space, and 'autonomy', 'achievement' and 'enjoyment' account for around 18% each. Across all attributes, greater value was placed on the difference between the lowest levels of capability than between the highest. This tariff will enable ICECAP-A to be used in economic evaluation both within the field of health and across public policy generally.

Measuring caregiver outcomes in palliative care: a construct validation study of two instruments for use in economic evaluations.

PURPOSE: Providing care to patients nearing the end of life can place a considerable burden on caregivers. Hence, policy decisions on interventions in palliative care should be guided by information on this burden. This study investigates construct validation of two preference-based caregiver outcome instruments suitable for economic evaluations: the Carer Experience Scale (CES) and the Care-related Quality of Life (CarerQol) instrument. Moreover, this study reports caregiver experiences in end-of-life care. METHODS: Data were collected with written questionnaires among caregivers of patients receiving palliative care services in the Southern metropolitan area of Adelaide, South Australia (n = 97). The effect of caregiving on caregivers was measured with the CES, CarerQol, Process Utility (PU) and Caregiver Strain Index (CSI). Convergent, discriminative and clinical validity were studied. RESULTS: As hypothesized, higher negative effect of caregiving measured on the CES was associated with higher negative effect on the CarerQol. Both the CES and CarerQol were associated in the expected positive direction with less strain from caregiving (CSI), more positive care experiences and more PU from caring. Caregivers' and care recipients' health status and duration of caregiving were negatively associated with caring experiences. CONCLUSIONS: Our findings suggest that the CES and CarerQol validly assess the effect of caregiving on caregivers in end-of-life care. Economic evaluations in end-of-life care should attempt to incorporate such instruments to provide a more holistic assessment of the true impact of interventions, especially where family and friends are heavily involved in caregiving.