Measurement properties of utility-based health-related quality of life measures in cardiac rehabilitation and secondary prevention programs: a systematic review.

PURPOSE: To identify utility-based patient-reported outcome measures (PROMs) for assessing health-related quality of life (HRQoL) in cardiac rehabilitation and secondary prevention programs (CR) and appraise existing evidence on their measurement properties. Secondly, to link their items to the International Classification of Functioning Disability and Health (ICF) and the International Consortium of Health Outcome Measures (ICHOM) domains for cardiovascular disease (CVD). METHODS: Eight databases were searched. The review followed the COSMIN and JBI guidelines for measurement properties systematic reviews and PRISMA 2020 reporting guidelines. Non-experimental and observational empirical studies of patients ≥ 18 years of age with CVD undergoing CR and assessed quality of life (QoL) or HRQoL using utility-based PROMs or one accompanied by health state utilities were included. RESULTS: Nine PROMs were identified with evidence on measurement properties for three measures: the German translations of SF-12, EQ-5D-5L, and MacNew heart disease HRQoL questionnaire. There was moderate quality evidence for responsiveness and hypothesis testing of the SF-12 and EQ-5D-5L, and high-quality evidence for responsiveness and hypothesis testing for the MacNew. All items of SF-12 and EQ-5D were linked to ICF categories, but four items of the MacNew were not classified or defined. All the PROM domains were mapped onto similar constructs from the ICHOM global sets. CONCLUSION: Three utility-based PROMs validated in CR were identified: the German versions of the EQ-5D and SF-12 and the MacNew questionnaire. These PROMs are linked to a breadth of ICF categories and all ICHOM global sets. Additional validation studies of PROMs in CR are required.

Responsiveness and construct validity of EPIC-26, AQoL-6D and SF-6D following treatment in prostate cancer.

PURPOSE: To assess construct validity and responsiveness of the Expanded Prostate Cancer Index Composite Instrument (EPIC-26) relative to the Short-Form Six-Dimension (SF-6D) and Assessment of Quality of Life 6-Dimension (AQoL-6D) in patients following treatment for prostate cancer. METHODS: Retrospective prostate cancer registry data were used. The SF-6D, AQoL-6D, and EPIC-26 were collected at baseline and one year post treatment. Analyses were based on Spearman's correlation coefficient, Bland-Altman plots and intra-class correlation coefficient, Kruskal Wallis, and Effect Size and the Standardised Response Mean for responsiveness. RESULTS: The study sample was comprised of 1915 patients. Complete case analysis of 3,697 observations showed moderate evidence of convergent validity between EPIC-26 vitality/hormonal domain and AQoL-6D (r = 0.45 and 0.54) and SF-6D (r = 0.52 and 0.56) at both timepoints. Vitality/hormonal domain also showed moderate convergent validity with coping domain of AQoL-6D (r = 0.45 and 0.54) and with role (r = 0.41 and 0.49) and social function (r = 0.47 and 0.50) domains of SF-6D at both timepoints, and with independent living (r = 0.40) and mental health (r = 0.43) of AQoL-6D at one year. EPIC-26 sexual domain had moderate convergent validity with relationship domain (r = 0.42 and 0.41) of AQoL-6D at both timepoints. Both AQoL-6D and SF-6D did not discriminate between age groups and tumour stage at both timepoints but AQoL-6D discriminated between outcomes for different treatments at one year. All EPIC-26 domains discriminated between age groups and treatment at both timepoints. The EPIC-26 was more responsive than AQoL-6D and SF-6D between baseline and one year following treatment. CONCLUSIONS: AQoL-6D can be used in combination with EPIC-26 in place of SF-12. Although EPIC-26 is not utility based, its popularity amongst clinicians and ability to discriminate between disease-specific characteristics and post-treatment outcomes in clinical trials makes it a candidate for use within cost-effectiveness analyses. The generic measure provides a holistic assessment of quality of life and is suitable for generating quality adjusted life years (QALYs).

What Constitutes Preferred Long-Term Care Provided in Residential Aged Care Facilities? An Empirical Comparison of the Preferences of the General Population, Residents, and Family Members.

OBJECTIVES: Relatively few studies to date have examined the preferences of members of the general population as potential future consumers of long-term aged care services. This study aimed to use discrete choice experiment methodology to compare the preferences of 3 groups: the general population, residents, and family members of people living in long-term aged care. METHODS: A total of 6 salient attributes describing the physical and psychosocial care in long-term residential aged care were drawn from qualitative research with people with a lived experience of aged care and were used to develop the discrete choice experiment questionnaire. The 6 attributes included: the level of time care staff spent with residents, homeliness of shared spaces, the homeliness of their own rooms, access to outside and gardens, frequency of meaningful activities, and flexibility with care routines. The questionnaire was administered to 1243 respondents including consumers (residents [n = 126], family member carers [n = 416]), and members of the general population (n = 701). RESULTS: For both the general population and resident samples, having their own room feeling "home-like" exhibited the largest impact upon overall preferences. For the family member sample, care staff being able to spend enough time exhibited the largest impact. Tests of poolability indicated that the resident and general population samples estimates could be pooled. The null hypothesis of equal parameters between the groups was rejected for the family members, indicating significant differences in preferences relative to the resident and the general population samples. CONCLUSIONS: This study illustrates that preferences for residential aged care delivery may vary depending upon perspective and experience.

A Systematic Review of International Guidance for Self-Report and Proxy Completion of Child-Specific Utility Instruments.

OBJECTIVES: This study aimed to identify and summarize published guidance and recommendations for child self- and proxy assessment of existing child-specific instruments of health-related quality of life (HRQoL) that are accompanied by utilities. METHODS: A total of 9 databases plus websites of (1) health technology assessment and health economics outcomes research organizations and (2) instrument developers were systematically searched. Studies were included if they reported guidance for child self- and proxy assessment for child populations (0-18 years old). Three reviewers independently screened titles, abstracts, and full-text reviews against the inclusion criteria. Key features of the guidance identified were summarized. RESULTS: A total of 19 studies met the inclusion criteria. In general, journal articles provided little guidance on child self- and proxy assessment, with the majority focused on instrument development and psychometric performance more broadly. Instrument developers' websites provided more guidance for child self- and proxy reports with specific guidance found for the EQ-5D-Y and the Pediatric Quality of Life Inventory. This guidance included the minimum age for self-report and mode of administration; recommended proxy types, age range of child for whom proxy report can be completed, and target population; and recall period. Websites of leading organizations provided general guidance on HRQoL evaluation in children but lacked specific guidance about self- and proxy completion. CONCLUSIONS: EQ-5D-Y and Pediatric Quality of Life Inventory developers' websites provided the most comprehensive guidance for self-report and proxy report of their respective instruments. More evidence is required for developing best practice guidance on why, when, and how to use self- and proxy reports in assessing HRQoL in child populations.

Cost-utility analysis of breast reduction surgery for women with symptomatic breast hypertrophy.

OBJECTIVE: To assess the cost-effectiveness of breast reduction surgery for women with symptomatic breast hypertrophy in Australia. DESIGN: Cost-utility analysis of data from a prospective cohort study. SETTING, PARTICIPANTS: Adult women with symptomatic breast hypertrophy assessed for bilateral breast reduction at the Flinders Medical Centre, a public tertiary hospital in Adelaide, April 2007 - February 2018. The control group included women with breast hypertrophy who had not undergone surgery. MAIN OUTCOME MEASURES: Health care costs (for the surgical admission and other related hospital costs within 12 months of surgery) and SF-6D utility scores (measure of health-related quality of life) were used to calculate incremental costs per quality-adjusted life-year (QALY) gained over 12 months, extrapolated to a 10-year time horizon. RESULTS: Of 251 women who underwent breast reduction, 209 completed the baseline and at least one post-operation assessment (83%; intervention group); 124 of 350 invited women waiting for breast reduction surgery completed the baseline and 12-month assessments (35%; control group). In the intervention group, the mean SF-6D utility score increased from 0.313 (SD, 0.263) at baseline to 0.626 (SD, 0.277) at 12 months; in the control group, it declined from 0.296 (SD, 0.267) to 0.270 (SD, 0.257). The mean QALY gain was consequently greater for the intervention group (adjusted difference, 1.519; 95% CI, 1.362-1.675). The mean hospital cost per patient was $11 857 (SD, $4322), and the incremental cost-effectiveness ratio (ICER) for the intervention was $7808 per QALY gained. The probability of breast reduction surgery being cost-effective was 100% at a willingness-to-pay threshold of $50 000 per QALY and 88% at $28 033 per QALY. CONCLUSIONS: Breast reduction surgery for women with symptomatic breast hypertrophy is cost-effective and should be available to women through the Australian public healthcare system.

Assessing the relative importance of key quality of life dimensions for people with and without a disability: an empirical ranking comparison study.

BACKGROUND: In economic evaluation, the quality of life of people with a disability has traditionally been assessed using preference-based instruments designed to measure and value quality of life. To provide robust measurement of the effectiveness of programs designed to improve the quality of life of people living with a disability, preference-based measures need to be sufficiently sensitive to detect incremental changes in the quality of life dimensions that are most important to people who have a disability. This study sought to explore whether there was a difference in the ranked order of importance of quality of life dimensions between people with a disability and people without a disability. METHODS: An online survey was developed and administered Australia wide. The first sample (n = 410) comprised adults (aged ≥ 18 years) with a disability (n = 208) and family carers of person/s with a disability who were asked to respond on behalf of the person with a disability (n = 202). The second sample included adults without disability (n = 443). Respondents were asked to rank the importance of 12 quality of life dimensions extracted from the content of established preference-based quality of life measures (EQ-5D, AQoL and ASCOT). RESULTS: People with a disability placed relatively higher importance on broader quality of life dimensions (e.g. Control, Independence, Self-care) relative to health status focused dimensions (e.g. Vision, Hearing, Physical mobility). This distinction was less differentiable for those 'without a disability'. The biggest differences in ranked importance of dimensions were in: Vision ('with disability' = 10th, 'without disability' = 4th), Self-care ('with disability' = 3rd, 'without disability' = 7th) and Mental well-being ('with disability' = 6th, 'without disability' = 2nd). CONCLUSIONS: The relative importance of quality of life dimensions for people with a disability differs to people without a disability. Quality of life is a key outcome for economic evaluation and for assessing the impact of disability care policy and practice in Australia and internationally. It is important that the effectiveness of interventions is measured and valued in ways which are fully reflective of the quality of life preferences of people with a disability.

Dietary sodium intake relates to vascular health in children with type 1 diabetes.

BACKGROUND AND OBJECTIVE: Children with type 1 diabetes (T1D) have vascular dysfunction and frequently struggle to adhere to dietary recommendations. Limited data exist for the vascular consequences of poor diet quality in children. We aimed to evaluate the association between dietary components and vascular function in children with T1D. METHODS: Cross-sectional study including 90 children (13.6 [3.5] years, 41 boys) with T1D. They had evaluation of dietary micro and macronutrients (Australian Child and Adolescent Eating Survey), vascular endothelial and smooth muscle function (flow-mediated dilatation and glyceryl trinitrate mediated dilatation [GTN], respectively), clinical and biochemical variables. RESULTS: Children had a sodium intake of 3.013 (0.76) (mean [SD]) g/day. Vascular smooth muscle dysfunction, as measured by GTN, related to higher daily sodium intake (r = -0.31, P = .003), independent of the inverse relationships between GTN and total energy (r = -0.30, P = .005) and fat intake (r = -0.28, P = .007). Multiregression model showed that an increase in 1 g of daily sodium intake was independently associated with a deterioration of 3 percentage units in GTN (95% CI -4.3, -0.9; P = .003). There was an association between sodium intake and systolic blood pressure after adjustment for age and gender (regression coefficient 2.4; 95% CI 0.5, 4.3; P = .01). CONCLUSIONS: High dietary sodium intake in children with T1D is common and relates to vascular dysfunction, independently of other dietary intake, blood pressure, and glycemic control.

A review of preference-based measures for the assessment of quality of life in children and adolescents with cerebral palsy.

PURPOSE: To examine the psychometric properties and suitability for use within the context of cerebral palsy research in children and adolescents of generic preference-based outcome measures (PROMs). METHODS: Nine electronic databases were searched in this systematic review. The consensus-based standards for the selection of health measurement instruments (COSMIN) checklist were used to measure the psychometric properties of the PROMs. A meta-analysis was used to pool correlation coefficients for convergent validity using the Schmidt-Hunter method. Heterogeneity was assessed using the I-squared statistic (I2). RESULTS: Four preference-based PROMs were identified from eight studies: Health Utilities Index-Mark 2 and 3 (HUI-2 and HUI-3, respectively), the Assessment Quality of Life-4 dimension (AQoL-4D) and the EuroQol-5 dimension 3 level (EQ-5D-3L). Only the HUI system was primarily developed for application with children/adolescents though health-state values for scoring the PROM were elicited from adults. The HUI-3 covered the most relevant constructs though it excludes important modules of health-related quality of life (HRQOL) such as activity limitations and participation restrictions. In terms of psychometric properties, evidence was presented for only five of COSMIN measurement properties: reliability (HUI3), measurement error (HUI-3), content validity (HUI-2 and HUI-3), Hypotheses testing (HUI-3 and AQoL-4D) and criterion validity (HUI-3). No papers reported on internal consistency, structural validity, cross-cultural validity or responsiveness of the preference-based measures in children and adolescents with cerebral palsy. CONCLUSIONS: This review highlights the dearth in studies using preference-based PROMs to measure HRQOL associated with cerebral palsy in children and adolescents. The HUI-3 demonstrated the strongest psychometric properties, though it does not cover all dimensions relevant to this population.

Suitability of the Epworth Sleepiness Scale (ESS) for Economic Evaluation: An Assessment of Its Convergent and Discriminant Validity.

OBJECTIVE: To assess the suitability for use within economic evaluation of a widely used sleep-related instrument (the Epworth Sleepiness Scale [ESS]) by examining its convergent and discriminant validity with two widely used generic preference-based instruments (Short-Form 36 [SF-36] and the Assessment of Quality of Life 4 dimensions [AQoL-4D]). METHODS: Data from a cross-section of 2,236 community-dwelling Australian men were analyzed. Convergent validity was investigated using Spearman's correlation, intraclass correlation, and modified Bland-Altman plots, while discriminant validity was examined using Kruskal Wallis tests. RESULTS: All instruments showed good discriminant validity. The ESS was weakly correlated to the Short Form 6 dimension, or SF-6D (derived from the SF-36) and AQoL-4D utilities (r = 0.20 and r = 0.19, respectively). Correlations between ESS and SF-36/AQoL-4D dimensions measuring the same construct were all in the hypothesized directions but also weak (range of absolute r = 0.00 to 0.18). The level of agreement between the ESS and AQoL-4D was the weakest, followed by that between the ESS and SF-6D. Moderate convergent validity was seen between the utilities. CONCLUSIONS: The lack of convergent validity between the ESS and the preference-based instruments shows that sleep-related constructs are not captured by the latter. The ESS has, however, demonstrated good discriminant validity comparable to that of the AQoL-4D and the SF-36/SF-6D and would therefore be equally useful for measuring subgroup differences within economic evaluation. We therefore recommend using the ESS within cost-effectiveness analysis as a complement to preference-based instruments in order to capture sleep-specific constructs not measured by the latter.

An equivalence evaluation of a nurse-moderated group-based internet support program for new mothers versus standard care: a pragmatic preference randomised controlled trial.

BACKGROUND: All mothers in South Australia are offered a clinic or home-visit by a Child and Family Health community nurse in the initial postnatal weeks. Subsequent support is available on request from staff in community clinics and from a telephone helpline. The aim of the present study is to compare equivalence of a single clinic-based appointment plus a nurse-moderated group-based internet intervention when infants were aged 0-6 months versus a single home-visit together with subsequent standard services (the latter support was available to mothers in both study groups). METHODS/DESIGN: The evaluation utilised a pragmatic preference randomised trial comparing the equivalence of outcomes for mothers and infants across the two study groups. Eligible mothers were those whose services were provided by nurses working in one of six community clinics in the metropolitan region of Adelaide. Mothers were excluded if they did not have internet access, required an interpreter, or their nurse clinician recommended that they not participate due to issues such as domestic violence or substance abuse. Randomisation was based on the service identification number sequentially assigned to infants when referred to the Child and Family Health Services from birthing units (this was done by administrative staff who had no involvement in recruiting mothers, delivering the intervention, or analyzing results for the study). Consistent with design and power calculations, 819 mothers were recruited to the trial. The primary outcomes for the trial are parents' sense of competence and self-efficacy measured using standard self-report questionnaires. Secondary outcomes include the quality of mother-infant relationships, maternal social support, role satisfaction and maternal mental health, infant social-emotional and language development, and patterns of service utilisation. Maternal and infant outcomes will be evaluated using age-appropriate questionnaires when infants are aged <2 months (pre-intervention), 9, 15, and 21 months. DISCUSSION: We know of no previous study that has evaluated an intervention that combines the capacity of nurse and internet-based services to improve outcomes for mothers and infants. The knowledge gained from this study will inform the design and conduct of community-based postnatal mother and child support programs. TRIAL REGISTRATION: Australian New Zealand Clinical Trials Registry ACTRN12613000204741.

An Investigation of Inter-Rater and Intra-Proxy Agreement in Measuring Quality of Life of Children in the Community Using the EQ-5D-Y-3L.

BACKGROUND: Self-reporting of health-related quality of life (HRQoL) in children is not always feasible. To date, proxy perspectives (Proxy versions 1 and 2) using the EQ-5D-Y-3L have not been explored for its impact on agreement with child self-report. Proxy version 1 requires the proxy to consider their own view of the child's HRQoL (proxy-proxy), while with Proxy version 2, the proxy is asked to respond as they believe their child would self-report their HRQoL (proxy-child). This study compared the inter-rater and intra-proxy agreement (overall and dimension level) using the EQ-5D-Y-3L self, proxy-proxy, and proxy-child reports. METHODS: A community-based sample of child (aged 6-12 years) and parent dyads were invited to participate in a semi-structured interview. The child self-completed the EQ-5D-Y-3L independently of the parent who completed the EQ-5D-Y-3L from proxy-proxy and proxy-child perspectives. Agreement was determined using Concordance Correlation Coefficients (CCCs) for the overall (preference-weighted) HRQoL, while agreement at the dimension level was evaluated using Gwet's agreement coefficient (AC1). To assess the differences between the self and the two proxy reports, the Wilcoxon matched-pair signed-rank test was used. RESULTS: This study involved 85 child-parent dyads. The agreement between self and proxy overall HRQoL was low (fair) with both proxy-proxy (CCC = 0.28) and proxy-child (CCC = 0.26) reports. The largest discrepancy in the child-proxy agreement at dimension level with both the proxy versions was observed for 'feeling worried, sad or unhappy'. Within this dimension, the proxy-child perspective resulted in a stronger agreement (AC1 = 0.7, good) with child self-report compared with the traditional proxy-proxy perspective (AC1 = 0.58, moderate). Although the preference-weighted HRQoL was consistent across both the proxy perspectives, a significant difference was observed in the EQ VAS scores (p = 0.02). CONCLUSIONS: This study demonstrates that choice of proxy perspective may have an impact on the problems reported on HRQoL dimensions and EQ VAS scores. However, in this community-based sample of generally healthy children, no significant difference was observed in the inter-rater agreement for child-self and proxy preference-weighted EQ-5D-Y-3L values based on proxy perspectives. While this suggests that preference-weighted data are not sensitive to the choice of perspective, these findings may differ for different HRQoL instruments and for alternative value sets with different properties.

Exploring the Use of Pictorial Approaches in the Development of Paediatric Patient-Reported Outcome Instruments: A Systematic Review.

INTRODUCTION: Children may find self-reporting health-related quality of life (HRQoL) using patient-reported outcome measures (PROMs) presented in text-based formats difficult, particularly younger children and children with developmental delays or chronic illness. In such cases, pictorial PROMs (where pictorial representations are used alongside or to replace text) may offer a valid alternative. AIM: This systematic literature review focused on identifying and describing paediatric PROMs that incorporate pictorial approaches, providing children with more effective means to express their HRQoL. METHODS: Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines were followed. Seven electronic databases were searched from inception to 1 March 2022. There were no country restrictions applied to the search; all English-language studies were considered for inclusion in the review. Characteristics and development methods of the identified pictorial PROMs were evaluated against context-specific good practice guidelines published by The Professional Society for Health Economics and Outcomes Research (ISPOR). RESULTS: A total of 22 paediatric pictorial PROMs, comprising 28 unique versions, were identified. These PROMs were predominantly developed in the USA and the UK, targeting children aged 3-18 years. Likert scales with pictorial anchors, particularly happy-sad faces, were commonly used for response options, appearing in 15 (54%) of the PROMs. Various graphic methods, such as happy-sad faces, cartoons, and thermometers, were adapted to specific content domains. These PROMs covered a wide range of domains, including physical and emotional health and social functioning. Emphasis was placed on content validity, including active child participation in developing pictorial elements. Notably, children's participation was sought during the development of the pictorial elements for 13 (46%) of the PROMs. Various development methods were employed, with 43% of paediatric PROMs using literature reviews, 43% using focus groups, and 32% involving expert consultation. Interviews emerged as the primary method, being employed in 61% of the studies. Additionally, three measures specifically addressed cross-cultural considerations. CONCLUSION: Paediatric pictorial PROMs offer child-friendly tools for assessing HRQoL for application with children who find reading and understanding text-based PROMs challenging. There is some evidence that pictorial PROMs facilitate self-report in this population and improve measurement properties compared to text-only PROMs. Further research is needed to develop, validate, and test paediatric pictorial PROMs, with an emphasis on including children from the inception in the co-design process.

Content Comparison of Quality-of-Life Instruments Used in Economic Evaluations of Sleep Disorder Interventions: A Systematic Review.

BACKGROUND: Assessment of quality of life (QoL) in people living with sleep disorders using questionnaires is necessary to compare intervention benefits. Knowledge of the content and concepts covered by specific QoL instruments is essential to determine which instruments are best suited for conducting economic evaluations of sleep-related interventions. OBJECTIVES: This review aims to identify the QoL instruments that have been applied in economic evaluations of sleep disorder interventions and compare their conceptual overlap and content coverage using the framework of the International Classification of Functioning, Disability and Health (ICF). METHODS: A systematic review of full economic evaluations in sleep published in peer-reviewed journals from conception to 30 May, 2023 was conducted. MEDLINE, PsychInfo, ProQuest, Cochrane, Scopus, CINAHL, Web of Science and Emcare were searched for eligible studies. Studies incorporating either generic or sleep-specific QoL instruments as the primary or secondary measures of effectiveness within a full economic evaluation were included. Quality appraisal against the JBI Critical Appraisal Checklist for Economic Evaluations and EURONHEED checklists and mapping of QoL items to ICF categories were performed by two reviewers, with a third helping settle any potential differences. RESULTS: Sixteen instruments were identified as having been used in sleep health economic evaluations. The EQ-5D-3L, Epworth Sleepiness Scale, and Insomnia Severity Index were the most widely used, but the latter two are predominantly diagnostic tools and not specifically designed to guide economic evaluations. Other instruments with broader ICF content coverage have been least used, and these include the Sleep Apnea Quality of Life Index, Functional Outcomes of Sleep Questionnaire, 15 Dimensions, Short-Form 6 Dimensions, 12-item Short Form Survey, 36-item Short Form Survey and the GRID Hamilton Rating Scale for Depression. CONCLUSIONS: This study provides an overview of current QoL instruments used in economic evaluations of sleep with respect to their content coverage. A combination of generic and sleep-specific instruments with broader ICF content coverage is recommended for such evaluations.

Child-Parent Agreement in the Assessment of Health-Related Quality of Life Using the CHU9D and the PedsQLTM.

OBJECTIVE: This study examined the inter-rater agreement between child-self and parental proxy health-related quality of life (HRQoL) ratings (overall and domain level) using two different generic child-specific measures, the Child Health Utility 9D (CHU9D) and the Pediatric Quality of Life Inventory (PedsQLTM), in a community-based sample of Australian children. A secondary objective was to investigate the impact of age on child-parent agreement across the dimensions of the two measures. METHODS: A total of 85 child-parent dyads (children aged 6-12 years) recruited from the community completed the self and proxy versions of the CHU9D and the PedsQLTM, respectively. The inter-rater agreement was estimated using Concordance Correlation Coefficients (CCC) and Gwet's Agreement Coefficient (AC1) for the overall sample and across age-groups. RESULTS: Agreement was low for overall HRQoL for both the CHU9D (CCC = 0.28) and the PedsQLTM (CCC = 0.39). Across the CHU9D dimensions, agreement was the highest for 'sad' (AC1 = 0.83) and lowest for 'tired' (AC1 = 0.31). The PedsQLTM demonstrated stronger agreement (AC1 = 0.41-0.6) for the physical health dimension but weaker for the psychosocial dimensions (AC1 < 0.4). Except for the 'tired' dimension, agreement was consistent across age-groups with the CHU9D, whilst the PedsQLTM showed poor agreement for most of the psychosocial health items among the older age-groups only (8-10 and 11-12 years). CONCLUSION: This study highlights that the agreement between child and parent proxy reported HRQoL may be influenced by both the measure used and the age of the child. These findings may have implications for the economic evaluation of healthcare interventions and services in child populations when both child and proxy perspectives are considered in the assessment of child HRQoL.

Measurement properties of utility-based health-related quality-of-life measures in cardiac rehabilitation: a systematic review protocol.

OBJECTIVE: This review will identify and appraise existing evidence on the measurement properties of utility-based health-related quality-of-life (HRQoL) measures used in cardiac rehabilitation programs. The review will map the measure domains against the International Classification of Functioning, Disability and Health and the International Consortium of Health Outcome Measures domains for cardiovascular disease. INTRODUCTION: Improving HRQoL is an international key indicator for delivering high-quality and person-centered secondary prevention programs. Many instruments and measures assess HRQoL in individuals undergoing cardiac rehabilitation. Utility-based measures are suitable for calculating quality-adjusted life years, a required outcome metric in cost-utility analysis. Cost-utility analysis requires the use of utility-based HRQoL measures. However, there is no consensus on which utility-based measure is best for populations undergoing cardiac rehabilitation. INCLUSION CRITERIA: Eligible studies will include patients aged ≥18 years with cardiovascular disease who are undergoing cardiac rehabilitation. Empirical studies that assess quality of life or HRQoL using a utility-based, health-related, patient-reported outcome measure or a measure accompanied by health state utilities will be eligible. Studies must report at least 1 of the following measurement properties: reliability, validity, responsiveness. METHODS: This review will follow the JBI methodology for systematic reviews of measurement properties. The following databases will be searched from inception to the present: MEDLINE, Emcare, Embase, Scopus, CINAHL, Web of Science Core Collection, Informit, PsyclNFO, REHABDATA, and the Cochrane Library. Studies will be critically appraised using the The COnsensus-based Standards for the selection of health status Measurement INstruments (COSMIN) risk of bias checklist. The review will be reported in line with the Preferred Reporting Items for Systematic reviews and Meta-Analysis (PRISMA) guidelines. REVIEW REGISTRATION: PROSPERO CRD42022349395.

Are We Agreed? Self- Versus Proxy-Reporting of Paediatric Health-Related Quality of Life (HRQoL) Using Generic Preference-Based Measures: A Systematic Review and Meta-Analysis.

OBJECTIVE: The aim of this study was to examine the level of agreement between self- and proxy-reporting of health-related quality of life (HRQoL) in children (under 18 years of age) using generic preference-based measures. METHODS: A systematic review of primary studies that reported agreement statistics for self and proxy assessments of overall and/or dimension-level paediatric HRQoL using generic preference-based measures was conducted. Where available, data on intraclass correlation coefficients (ICCs) were extracted to summarise overall agreement levels, and Cohen's kappa was used to describe agreement across domains. A meta-analysis was also performed to synthesise studies and estimate the level of agreement between self- and proxy-reported paediatric overall and domain-level HRQoL. RESULTS: Of the 30 studies included, 25 reported inter-rater agreement for overall utilities, while 17 reported domain-specific agreement. Seven generic preference-based measures were identified as having been applied: Health Utilities Index (HUI) Mark 2 and 3, EQ-5D measures, Child Health Utility 9 Dimensions (CHU9D), and the Quality of Well-Being (QWB) scale. A total of 45 dyad samples were included, with a total pooled sample of 3084 children and 3300 proxies. Most of the identified studies reported a poor inter-rater agreement for the overall HRQoL using ICCs. In contrast to more observable HRQoL domains relating to physical health and functioning, the inter-rater agreement was low for psychosocial-related domains, e.g., 'emotion' and 'cognition' attributes of both HUI2 and HUI3, and 'feeling worried, sad, or unhappy' and 'having pain or discomfort' domains of the EQ-5D. Parents demonstrated a higher level of agreement with children relative to health professionals. Child self- and proxy-reports of HRQoL showed lower agreement in cancer-related studies than in non-cancer-related studies. The overall ICC from the meta-analysis was estimated to be 0.49 (95% confidence interval 0.34-0.61) with poor inter-rater agreement. CONCLUSION: This study provides evidence from a systematic review of studies reporting dyad assessments to demonstrate the discrepancies in inter-rater agreement between child and proxy reporting of overall and domain-level paediatric HRQoL using generic preference-based measures. Further research to drive the inclusion of children in self-reporting their own HRQoL wherever possible and limiting the reliance on proxy reporting of children's HRQoL is warranted.

Convergent and Discriminant Validity of the Barthel Index and the EQ-5D-3L When Used on Older People in a Rehabilitation Setting.

This study compares the empirical performance of a commonly used functional-status measure, the Barthel Index (BI), to that of a widely used generic preference-based instrument, the EuroQoL-5-Dimensions 3 Level (EQ-5D-3L), in older people. Data from older people receiving rehabilitation services were used to test the validity of the BI and EQ-5D-3L. Convergent validity was investigated using Spearman's correlation, exploratory factor analysis (EFA), scatter plots, Krippendorff's alpha and modified Bland-Altman plots. Discriminant validity was examined using Kruskal Wallis tests, ceiling effects and EFA. A total of 1690 participants were included in the analysis. The BI total and EQ-5D-3L utility scores showed moderate correlation (r = 0.51; Krippendorff's alpha = 0.52). Kendall's Tau-B correlations between BI items and EQ-5D-3L dimensions measuring the same construct were weak to moderate (0.05 ≤ absolute r ≤ 0.54). In the EFA, some BI items cross-loaded onto the same factors as EQ-5D-3L dimensions, suggesting that the instruments were interrelated. The BI, however, focuses more on physical functioning, while the EQ-5D-3L measures broader wellbeing concepts. Both instruments showed good discriminant validity and would therefore be equally valuable for measuring subgroup differences. Researchers should consider using the BI in rehabilitation to capture more physical functioning-specific constructs not measured by the EQ-5D-3L.

International guidelines for self-report and proxy completion of paediatric health-related quality of life measures: a protocol for a systematic review.

INTRODUCTION: Measures of health-related quality of life (HRQoL), accompanied by the values (or utilities) required to estimate quality-adjusted life-years, are crucial for determining health benefits within economic evaluation and health technology assessment. Several generic and condition-specific measures or instruments of HRQoL, accompanied by values, currently exist for application with child populations. However, there is a lack of a structured summary of guidelines and recommendations for applying these measures in practice. This protocol describes a systematic review of guidelines and recommendations for child and proxy completion of child-specific measures of HRQoL. The aims of the review are to (1) identify and summarise published guidelines and recommendations for existing child-specific measures of HRQoL, (2) determine whether the identified guidelines and recommendations differ by instrument and child characteristics, (3) identify current gaps in these guidelines and recommendations and (4) identify best practices for child self and proxy assessment in paediatric HRQoL measurement for economic evaluation and health technology assessment. METHODS AND ANALYSIS: The review will identify, collate and synthesise published guidelines and recommendations for existing child-specific utility measures of HRQoL. Electronic databases to be searched include the Cochrane Library, Medline, Scopus, Web of Science, EconLit, PsycINFO, CINAHL, Embase and Informit. The search will be extended to websites of (1) international organisations for health technology assessment, (2) regulation, health economics and HRQoL outcomes research and (3) instrument developers. Three reviewers will independently screen titles and abstracts against the inclusion criteria. A narrative synthesis will describe the key features of the guidelines identified. ETHICS AND DISSEMINATION: Ethical approval is not required as the proposed systematic review will not use primary data. A paper of the systematic review will be submitted to a peer-reviewed journal for publication. PROSPERO REGISTRATION NUMBER: CRD42020207160.

A preference for quality: Australian general public's willingness to pay for home and residential aged care.

In Australia and many other countries internationally, aged care services are provided to older people in their own homes or residential care facilities. The majority of these services are funded by the federal government using taxpayer contributions from the general public. However, the monetary value Australians place on aged care services, and the factors that predict this value, have not been examined. We, therefore, sought to determine the general public's willingness to pay (WTP) for aged care services and examine which factors influence this WTP. A cross-sectional contingent valuation survey was administered to a nationally representative cohort of 10,285 Australians between September and October 2020 from the general population aged 18 years and over. Respondents were asked to indicate their WTP values for satisfactory and high-quality aged care services to be provided in the future. A two-part regression model was used to explain what factors explained variation in WTP. In total, 80% (61%) of respondents were willing to pay to access satisfactory (high) quality home care (counterpart figures for residential care were 64% (45%)). On average, respondents were willing to pay between $126 and $158 ($145 and $237) per week to receive satisfactory-quality (high-quality) home care and between $333 and $520 ($308 and $680) per week for satisfactory-quality (high-quality) residential care. Respondents were willing to pay an additional $120 per week on average to access high-quality aged care. Higher WTP values were generally associated with being younger, male, recent experience with aged care through a close family member accessing aged care and ability to pay. These results suggest general public support for payment of individual co-contributions to access aged care services in the future.

Mapping the PedsQL™ onto the CHU9D: An Assessment of External Validity in a Large Community-Based Sample.

BACKGROUND: Mapping algorithms have been indicated as a second-best solution for estimating health state utilities for the calculation of quality-adjusted life-years within cost-utility analysis when no generic preference-based measure is incorporated into the study. However, the predictive performance of these algorithms may be variable and hence it is important to assess their external validity before application in different settings. OBJECTIVE: The aim of this study was to assess the external validity and generalisability of existing mapping algorithms for predicting preference-based Child Health Utility 9D (CHU9D) utilities from non-preference-based Pediatric Quality of Life Inventory (PedsQL) scores among children and adolescents living with or without disabilities or health conditions. METHODS: Five existing mapping algorithms, three developed using data from an Australian community population and two using data from a UK population with one or more self-reported health conditions, were externally validated on data from the Longitudinal Study of Australian Children (n = 6623). The predictive accuracy of each mapping algorithm was assessed using the mean absolute error (MAE) and the mean squared error (MSE). RESULTS: Values for the MAE (0.0741-0.2302) for all validations were within the range of published estimates. In general, across all ages, the algorithms amongst children and adolescents with disabilities/health conditions (Australia MAE: 0.2085-0.2302; UK MAE: 0.0854-0.1162) performed worse relative to those amongst children and adolescents without disabilities/health conditions (Australia MAE: 0.1424-0.1645; UK MAE: 0.0741-0.0931). CONCLUSIONS: The published mapping algorithms have acceptable predictive accuracy as measured by MAE and MSE. The findings of this study indicate that the choice of the most appropriate mapping algorithm to apply may vary according to the population under consideration.