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Objectives: Pediatric Quality-of-Life Inventory Version 4.0 Generic Core Scales (PedsQL GCS), comprising 23 items covering 4 subscales (physical, emotional, social, and school functioning), is a widely applied generic measure of childhood health-related quality of life but does not provide health utilities for cost-effectiveness-based decision making. This study aimed to develop a reduced item version of PedsQL GCS amenable to health utility derivation in Australia., Methods: Data sources were 2 cohorts of the Longitudinal Study of Australian Children, including proxy responses for all PedsQL GCS versions (Toddlers, Young Children, Children, and Teens), and the CheckPoint sample containing child self-report to the Children version. Three analytic samples were CheckPoint sample (n = 1874); Mallinson sample containing 1 measurement per child from one of the Young Children, Children, or Teens versions (n = 7855); and Toddlers sample (n = 7401). Exploratory and confirmatory factor analyses assessed dimensionality. Psychometric analyses used Rasch and classical criteria on 3 randomly selected subsamples (n = 500) per sample. Item selection prioritized psychometric performance in the CheckPoint sample, also considering performance in other samples and conceptual content., Results: Dimensionality assessments did not generate an alternative empirical structure for the measure, and psychometric analyses were conducted on the original 4 subscales. The selected items were: "Get aches and pains" for physical functioning; "Feel sad/blue" for emotional functioning; "Other kids not friends" for social functioning; and "Keeping up with school work" for school functioning., Conclusions: The final 4-item set, pending further psychometric validation and valuation, can generate health utilities from the widely used PedsQL GCS to inform cost-effectiveness-based decision making., Competing Interests: Author Disclosures Author disclosure forms can be accessed below in the Supplemental Material section. Dr Petrou is an editor for Value in Health and had no role in the peer-review process of this article., (Copyright © 2025. Published by Elsevier Inc.)

Background: Recent systematic reviews show varying methods for eliciting, modelling, and reporting preference-based values for child health-related quality-of-life (HRQoL) outcomes, thus producing value sets with different characteristics. Reporting in many of the reviewed studies was found to be incomplete and inconsistent, making them difficult to assess. Checklists can help to improve standards of reporting; however, existing checklists do not address methodological issues for valuing child HRQoL. Existing checklists also focus on reporting methods and processes used in developing HRQoL values, with less focus on reporting of the values' key characteristics and properties. We aimed to develop a checklist for studies generating values for child HRQoL, including for disease-specific states and value sets for generic child HRQoL instruments., Development: A conceptual model provided a structure for grouping items into five modules. Potential items were sourced from an adult HRQoL checklist review, with additional items specific to children developed using recent reviews. Checklist items were reduced by eliminating duplication and overlap, then refined for relevance and clarity via an iterative process. Long and short checklist versions were produced for different user needs. The resulting long RETRIEVE contains 83 items, with modules for reporting methods (A-D) and characteristics of values (E), for researchers planning and reporting child health valuation studies. The short RETRIEVE contains 14 items for decision makers or researchers choosing value sets., Conclusion: Applying the RETRIEVE checklists to relevant studies suggests feasibility. RETRIEVE has the potential to improve completeness in the reporting of preference-based values for child HRQOL outcomes and to improve assessment of preference-based value sets., (© 2024. The Author(s).)

Objectives: We aimed to synthesise knowledge on the relative social value of child and adult health., Methods: Quantitative and qualitative studies that evaluated the willingness of the public to prioritise treatments for children over adults were included. A search to September 2023 was undertaken. Completeness of reporting was assessed using a checklist derived from Johnston et al. Findings were tabulated by study type (matching/person trade-off, discrete choice experiment, willingness to pay, opinion survey or qualitative). Evidence in favour of children was considered in total, by length or quality of life, methodology and respondent characteristics., Results: Eighty-eight studies were included; willingness to pay (n = 9), matching/person trade-off (n = 12), discrete choice experiments (n = 29), opinion surveys (n = 22) and qualitative (n = 16), with one study simultaneously included as an opinion survey. From 88 studies, 81 results could be ascertained. Across all studies irrespective of method or other characteristics, 42 findings supported prioritising children, while 12 provided evidence favouring adults in preference to children. The remainder supported equal prioritisation or found diverse or unclear views. Of those studies considering prioritisation within the under 18 years of age group, nine findings favoured older children over younger children (including for life saving interventions), six favoured younger children and five found diverse views., Conclusions: The balance of evidence suggests the general public favours prioritising children over adults, but this view was not found across all studies. There are research gaps in understanding the public's views on the value of health gains to very young children and the motivation behind the public's views on the value of child relative to adult health gains., Clinical Trial Registration: The review is registered at PROSPERO number: CRD42021244593. There were two amendments to the protocol: (1) some additional search terms were added to the search strategy prior to screening to ensure coverage and (2) a more formal quality assessment was added to the process at the data extraction stage. This assessment had not been identified at the protocol writing stage., (© 2023. The Author(s).)

Background: Understanding the physical and mental health needs of the population through evidence-based research is a priority for informing health policy. During the COVID-19 pandemic, population wellbeing dramatically dropped. The relationship between experiences of symptomatic illness episodes and health-related quality of life has been less documented., Objective: This study analysed the association between symptomatic COVID-19 illness and health-related quality of life., Methods: The analyses drew from a cross-sectional analysis of data from a national digital symptoms' surveillance survey conducted in the UK in 2020. We identified illness episodes using symptoms and test results data and we analysed validated health-related quality of life outcomes including health utility scores (indexed on a 0-1 cardinal scale) and visual analogue scale (VAS) scores (0-100 scale) generated by the EuroQoL's EQ-5D-5L measure. The econometric model controlled for respondents' demographic and socioeconomic characteristics, comorbidities, social isolation measures, and regional and time fixed effects., Results: The results showed that the experience of common SARS-CoV-2 symptoms was significantly associated with poorer health-related quality of life across all EQ-5D-5L dimensions of mobility, self-care, usual activities, pain/discomfort and anxiety/depression, a decrement in utility score of - 0.13 and a decrement in the EQ-VAS score of - 15. The findings were robust to sensitivity analyses and restrictive test results-based definitions., Conclusion: This evidence-based study highlights the need for targeting of interventions and services towards those experiencing symptomatic episodes during future waves of the pandemic and helps to quantify the benefits of SARS-CoV-2 treatment in terms of health-related quality of life., (© 2023. The Author(s), under exclusive licence to Springer Nature Switzerland AG.)

Background: Childhood multi-attribute utility instruments (MAUIs) can be used to measure health utilities in children (aged ≤ 18 years) for economic evaluation. Systematic review methods can generate a psychometric evidence base that informs their selection for application. Previous reviews focused on limited sets of MAUIs and psychometric properties, and only on evidence from studies that directly aimed to conduct psychometric assessments., Objective: This study aimed to conduct a systematic review of psychometric evidence for generic childhood MAUIs and to meet three objectives: (1) create a comprehensive catalogue of evaluated psychometric evidence; (2) identify psychometric evidence gaps; and (3) summarise the psychometric assessment methods and performance by property., Methods: A review protocol was registered with the Prospective Register of Systematic Reviews (PROSPERO; CRD42021295959); reporting followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) 2020 guideline. The searches covered seven academic databases, and included studies that provided psychometric evidence for one or more of the following generic childhood MAUIs designed to be accompanied by a preference-based value set (any language version): 16D, 17D, AHUM, AQoL-6D, CH-6D, CHSCS-PS, CHU9D, EQ-5D-Y-3L, EQ-5D-Y-5L, HUI2, HUI3, IQI, QWB, and TANDI; used data derived from general and/or clinical childhood populations and from children and/or proxy respondents; and were published in English. The review included 'direct studies' that aimed to assess psychometric properties and 'indirect studies' that generated psychometric evidence without this explicit aim. Eighteen properties were evaluated using a four-part criteria rating developed from established standards in the literature. Data syntheses identified psychometric evidence gaps and summarised the psychometric assessment methods/results by property., Results: Overall, 372 studies were included, generating a catalogue of 2153 criteria rating outputs across 14 instruments covering all properties except predictive validity. The number of outputs varied markedly by instrument and property, ranging from 1 for IQI to 623 for HUI3, and from zero for predictive validity to 500 for known-group validity. The more recently developed instruments targeting preschool children (CHSCS-PS, IQI, TANDI) have greater evidence gaps (lack of any evidence) than longer established instruments such as EQ-5D-Y, HUI2/3, and CHU9D. The gaps were prominent for reliability (test-retest, inter-proxy-rater, inter-modal, internal consistency) and proxy-child agreement. The inclusion of indirect studies (n = 209 studies; n = 900 outputs) increased the number of properties with at least one output of acceptable performance. Common methodological issues in psychometric assessment were identified, e.g., lack of reference measures to help interpret associations and changes. No instrument consistently outperformed others across all properties., Conclusion: This review provides comprehensive evidence on the psychometric performance of generic childhood MAUIs. It assists analysts involved in cost-effectiveness-based evaluation to select instruments based on the application-specific minimum standards of scientific rigour. The identified evidence gaps and methodological issues also motivate and inform future psychometric studies and their methods, particularly those assessing reliability, proxy-child agreement, and MAUIs targeting preschool children., (© 2023. The Author(s).)

Background: The Paediatric Quality of life Inventory (PedsQL TM ) Generic Core Scales and the Child Health Utilities 9 Dimensions (CHU9D) are two paediatric health-related quality of life (HRQoL) measures commonly used in overweight and obesity research. However, no studies have comprehensively established the psychometric properties of these instruments in the context of paediatric overweight and obesity. The aim of this study was to assess the reliability, acceptability, validity and responsiveness of the PedsQL and the CHU9D in the measurement of HRQoL among children and adolescents living with overweight and obesity., Subjects/methods: Subjects were 6544 child participants of the Longitudinal Study of Australian Children, with up to 3 repeated measures of PedsQL and CHU9D and aged between 10 and 17 years. Weight and height were measured objectively by trained operators, and weight status determined using World Health Organisation growth standards. We examined reliability, acceptability, known group and convergent validity and responsiveness, using recognised methods., Results: Both PedsQL and CHU9D demonstrated good internal consistency reliability, and high acceptability. Neither instrument showed strong convergent validity, but PedsQL appears to be superior to the CHU9D in known groups validity and responsiveness. Compared with healthy weight, mean (95%CI) differences in PedsQL scores for children with obesity were: boys -5.6 (-6.2, -4.4); girls -6.7 (-8.1, -5.4) and differences in CHU9D utility were: boys -0.02 (-0.034, -0.006); girls -0.035 (-0.054, -0.015). Differences in scores for overweight compared with healthy weight were: PedsQL boys -2.2 (-3.0, -1.4) and girls -1.3 (-2.0, -0.6) and CHU9D boys: no significant difference; girls -0.014 (-0.026, -0.003)., Conclusion: PedsQL and CHU9D overall demonstrated good psychometric properties, supporting their use in measuring HRQoL in paediatric overweight and obesity. CHU9D had poorer responsiveness and did not discriminate between overweight and healthy weight in boys, which may limit its use in economic evaluation., (© 2023. The Author(s).)

Background: The most appropriate preference-based health-related quality of life (HRQoL) instruments for trials or research studies that ascertain the consequences of individuals born very preterm and/or low birthweight (VP/VLBW) are not known. Agreement between the HUI3 and SF-6D multi-attribute utility measures have not been previously investigated for VP/VLBW and normal birthweight or term-born controls. This study examined the agreement between the outputs of the HUI3 and SF-6D measures among adults born VP/VLBW and normal birthweight or term born controls., Methods: We used two prospective cohorts of individuals born VP/VLBW and controls contributing to the 'Research on European Children and Adults Born Preterm' (RECAP) consortium which assessed HRQoL using two preference-based measures. The combined dataset of individual participant data (IPD) included 407 adult VP/VLBW survivors and 367 controls, ranging in age from 18 to 26 years. Bland-Altman plots, intra-class correlation coefficients, and generalized linear mixed models in a one-step approach were used to examine agreement between the measures., Results: There was significant discordance between the HUI3 and SF-6D multi-attribute utility measures in the VP/VLBW sample, controls, and in the combined samples. Agreement between the HUI3 and SF-6D multi-attribute utility measures was weaker in controls compared with VP/VLBW individuals., Conclusions and Relevance: The HUI3 and SF-6D each provide unique information on different aspects of health status across the groups. The HUI3 better captures preterm-related changes to HRQoL in adulthood compared to SF-6D. Studies focused on measuring physical or cognitive aspects of health will likely benefit from using the HUI3 instead of the SF-6D, regardless of gestational age at birth and birthweight status., (© 2023. The Author(s).)

Objectives: The EuroQol Group has developed an extended version of the EQ-5D-Y-3L with five response levels for each of its five dimensions (EQ-5D-Y-5L). The psychometric performance has been reported in several studies for the EQ-5D-Y-3L but not for the EQ-5D-Y-5L. This study aimed to psychometrically evaluate the EQ-5D-Y-3L and EQ-5D-Y-5L Chichewa (Malawi) versions., Methods: The EQ-5D-Y-3L, EQ-5D-Y-5L and PedsQL™ 4.0 Chichewa versions were administered to children and adolescents aged 8-17 years in Blantyre, Malawi. Both of the EQ-5D-Y versions were evaluated for missing data, floor/ceiling effects, and validity (convergent, discriminant, known-group and empirical)., Results: A total of 289 participants (95 healthy, and 194 chronic and acute) self-completed the questionnaires. There was little problem with missing data (< 5%) except in children aged 8-12 years particularly for the EQ-5D-Y-5L. Ceiling effects was generally reduced in moving from the EQ-5D-Y-3L to the EQ-5D-Y-5L. For both EQ-5D-Y-3L and EQ-5D-Y-5L, convergent validity tested with PedsQL™ 4.0 was found to be satisfactory (correlation ≥ 0.4) at scale level but mixed at dimension /sub-scale level. There was evidence of discriminant validity (p > 0.05) with respect to gender and age, but not for school grade (p < 0.05). For empirical validity, the EQ-5D-Y-5L was 31-91% less efficient than the EQ-5D-Y-3L at detecting differences in health status using external measures., Conclusions: Both versions of the EQ-5D-Y-3L and EQ-5D-Y-5L had issues with missing data in younger children. Convergent validity, discriminant validity with respect to gender and age, and known-group validity of either measures were also met for use among children and adolescents in this population, although with some limitations (discriminant validity by grade and empirical validity). The EQ-5D-Y-3L seems particularly suited for use in younger children (8-12 years) and the EQ-5D-Y-5L in adolescents (13-17 years). However, further psychometric testing is required for test re-test reliability and responsiveness that could not be carried out in this study due to COVID-19 restrictions., (© 2023. The Author(s).)

Background and Objective: Assessment of health-related quality of life for individuals born very preterm and/or low birthweight (VP/VLBW) offers valuable complementary information alongside biomedical assessments. However, the impact of VP/VLBW status on health-related quality of life in adulthood is inconclusive. The objective of this study was to examine associations between VP/VLBW status and preference-based health-related quality-of-life outcomes in early adulthood., Methods: Individual participant data were obtained from five prospective cohorts of individuals born VP/VLBW and controls contributing to the 'Research on European Children and Adults Born Preterm' Consortium. The combined dataset included over 2100 adult VP/VLBW survivors with an age range of 18-29 years. The main exposure was defined as birth before 32 weeks' gestation (VP) and/or birth weight below 1500 g (VLBW). Outcome measures included multi-attribute utility scores generated by the Health Utilities Index Mark 3 and the Short Form 6D. Data were analysed using generalised linear mixed models in a one-step approach using fixed-effects and random-effects models., Results: VP/VLBW status was associated with a significant difference in the Health Utilities Index Mark 3 multi-attribute utility score of - 0.06 (95% confidence interval - 0.08, - 0.04) in comparison to birth at term or at normal birthweight; this was not replicated for the Short Form 6D. Impacted functional domains included vision, ambulation, dexterity and cognition. VP/VLBW status was not associated with poorer emotional or social functioning, or increased pain., Conclusions: VP/VLBW status is associated with lower overall health-related quality of life in early adulthood, particularly in terms of physical and cognitive functioning. Further studies that estimate the effects of VP/VLBW status on health-related quality-of-life outcomes in mid and late adulthood are needed., (© 2022. The Author(s).)

Background: The Headache Impact Test (HIT-6) and the Chronic Headache Questionnaire (CH-QLQ) measure headache-related quality of life but are not preference-based and therefore cannot be used to generate health utilities for cost-effectiveness analyses. There are currently no established algorithms for mapping between the HIT-6 or CH-QLQ and preference-based health-related quality-of-life measures for chronic headache population., Methods: We developed algorithms for generating EQ-5D-5L and SF-6D utilities from the HIT-6 and the CHQLQ using both direct and response mapping approaches. A multi-stage model selection process was used to assess the predictive accuracy of the models. The estimated mapping algorithms were derived to generate UK tariffs and was validated using the Chronic Headache Education and Self-management Study (CHESS) trial dataset., Results: Several models were developed that reasonably accurately predict health utilities in this context. The best performing model for predicting EQ-5D-5L utility scores from the HIT-6 scores was a Censored Least Absolute Deviations (CLAD) (1) model that only included the HIT-6 score as the covariate (mean squared error (MSE) 0.0550). The selected model for CH-QLQ to EQ-5D-5L was the CLAD (3) model that included CH-QLQ summary scores, age, and gender, squared terms and interaction terms as covariates (MSE 0.0583). The best performing model for predicting SF-6D utility scores from the HIT-6 scores was the CLAD (2) model that included the HIT-6 score and age and gender as covariates (MSE 0.0102). The selected model for CH-QLQ to SF-6D was the OLS (2) model that included CH-QLQ summary scores, age, and gender as covariates (MSE 0.0086)., Conclusion: The developed algorithms enable the estimation of EQ-5D-5L and SF-6D utilities from two headache-specific questionnaires where preference-based health-related quality of life data are missing. However, further work is needed to help define the best approach to measuring health utilities in headache studies., (© 2022. The Author(s).)

Background: Quality of life (QoL) is an important measure of disease burden and general health perception. The relationship between early chronic kidney disease (CKD) and QoL remains poorly understood. The Oxford Renal Study (OxRen) cohort comprises 1063 adults aged ≥60 years from UK primary care practices screened for early CKD, grouped according to existing or screen-detected CKD diagnoses, or biochemistry results indicative of reduced renal function (referred to as transient estimated glomerular filtration rate (eGFR) reduction)., Objectives: This study aimed to compare QoL in participants known to have CKD at recruitment to those identified as having CKD through a screening programme., Methods: Health profile data and multi-attribute utility scores were reported for two generic questionnaires: 5-level EuroQol-5 Dimension (EQ-5D-5L) and ICEpop CAPability measure for Adults (ICECAP-A). QoL was compared between patients with existing and screen-detected CKD; those with transient eGFR reduction served as the reference group in univariable and multivariable linear regression., Results: Mean and standard deviation utility scores were not significantly different between the subgroups for EQ-5D-5L (screen-detected:0.785±0.156, n = 480, transient:0.779±0.157, n = 261, existing CKD:0.763±0.171, n = 322, p = 0.216) or ICECAP-A (screen-detected:0.909±0.094, transient:0.904±0.110, existing CKD:0.894±0.115, p = 0.200). Age, smoking status, and number of comorbidities were identified as independent predictors of QoL in this cohort., Conclusion: QoL of participants with existing CKD diagnoses was not significantly different from those with screen-detected CKD or transient eGFR reduction and was similar to UK mean scores for the same age, suggesting that patient burden of early CKD is minor. Moreover, CKD-related comorbidities contribute more significantly to disease burden in earlier stages of CKD than renal function per se. Larger prospective studies are required to define the relationship between QoL and CKD progression more precisely. These data also confirm the essentially asymptomatic nature of CKD, implying that routine screening or case finding are required to diagnose it., Competing Interests: I have read the journal’s policy and the authors of this manuscript have the following competing interests: FDRH reports fees or grants from Novartis, fees or other from Boehringer Ingelheim and Pfizer outside the submitted work. This does not alter our adherence to PLOS ONE policies on sharing data and materials. All other authors have no conflict of interest to declare.

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., (© 2022. The Author(s).)

Background and Objectives: Valuing children's health states for use in economic evaluations is globally relevant and is of particular relevance in jurisdictions where a cost-utility analysis is the preferred form of analysis for decision making. Despite this, the challenges with valuing child health mean that there are many remaining questions for debate about the approach to elicitation of values. The aim of this paper was to identify and describe the methods used to value children's health states and the specific issues that arise in the use of these methods., Methods: We conducted a systematic search of electronic databases to identify studies published in English since 1990 that used preference elicitation methods to value child and adolescent (under 18 years of age) health states. Eligibility criteria comprised valuation studies concerning both child-specific patient-reported outcome measures and child health states defined in other ways, and methodological studies of valuation approaches that may or may not have yielded a value set algorithm., Results: A total of 77 eligible studies were identified from which data on country setting, aims, condition (general population or clinically specific), sample size, age of respondents, the perspective that participants were asked to adopt, source of values (respondents who completed the preference elicitation tasks) and methods questions asked were extracted. Extracted data were classified and evaluated using narrative synthesis methods. The studies were classified into three groups: (1) studies comparing elicitation methods (n = 30); (2) studies comparing perspectives (n = 23); and (3) studies where no comparisons were presented (n = 26); selected studies could fall into more than one group. Overall, the studies varied considerably both in methods used and in reporting. The preference elicitation tasks included time trade-off, standard gamble, visual analogue scaling, rating/ranking, discrete choice experiments, best-worst scaling and willingness to pay elicited through a contingent valuation. Perspectives included adults' considering the health states from their own perspective, adults taking the perspective of a child (own, other, hypothetical) and a child/adolescent taking their own or the perspective of another child. There was some evidence that children gave lower values for comparable health states than did adults that adopted their own perspective or adult/parents that adopted the perspective of children., Conclusions: Differences in reporting limited the conclusions that can be formed about which methods are most suitable for eliciting preferences for children's health and the influence of differing perspectives and values. Difficulties encountered in drawing conclusions from the data (such as lack of consensus and poor reporting making it difficult for users to choose and interpret available values) suggest that reporting guidelines are required to improve the consistency and quality of reporting of studies that value children's health using preference-based techniques., (© 2022. The Author(s).)

Objectives: The EuroQol Group is developing a new EQ-5D-Y-5L version with 5 severity levels for each of the 5 dimensions. The 5 severity levels describe different health severities and there is a potential for severity level inversion. This article aims to report the process of cross-cultural adaptation of the beta EQ-5D-Y-5L into Chichewa (Malawi) using the card ranking exercise, which has been added to the EQ-5D-Y-5L translation protocol., Methods: To assess the correct hierarchical ordering of severity levels, the adaptation followed the EQ-5D-Y-5L translation protocol. Cognitive interviews were undertaken to establish conceptual equivalence. Thereafter, 4 iterations of ranking exercises were conducted, leading to amendments of the translated Chichewa version to arrive at a final version., Results: The iterations were assessed by 18 participants aged 8 to 14 years. Health proved to be a difficult concept to translate as was "discomfort." Cognitive interviews identified further conceptual issues, particularly with the "looking after myself" dimension. Considerations about lack of soap or water indicated that some children did not fully comprehend this dimension as being about the ability to wash and dress themselves. The iterative card ranking exercise detected severity level inversion between "a little bit" and "some," and between "a lot" and "extreme" and alternative Chichewa words/phrases were then tested. Ultimately, the intended hierarchical severity ranking was achieved and an acceptable Chichewa version was produced., Conclusions: Conceptual and linguistic equivalence to the English EQ-5D-Y-5L was established for the Chichewa EQ-5D-Y-5L version. The card ranking exercise was instrumental in correcting severity level inversion and supporting the comprehensible translation., (Copyright © 2021 ISPOR--The professional society for health economics and outcomes research. Published by Elsevier Inc. All rights reserved.)

Background and Aims: Patient-reported outcome measures (PROMs) for children (aged ≤ 18 years) present methodological challenges. PROMs can be categorised by their diverse underlying conceptual bases, including functional, disability and health (FDH) status; quality of life (QoL); and health-related quality of life (HRQoL). Some PROMs are designed to be accompanied by preference weights. PROMs should account for childhood developmental differences by incorporating age-appropriate health/QoL domains, guidance on respondent type(s) and design. This systematic review aims to identify generic multidimensional childhood PROMs and synthesise their characteristics by conceptual basis, target age, measurement considerations, and the preference-based value sets that accompany them., Methods: The study protocol was registered in the Prospective Register of Systematic Reviews (CRD42021230833), and reporting followed Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. We conducted systematic database searches for generic multidimensional childhood PROMs covering the period 2012-2020, which we combined with published PROMs identified by an earlier systematic review that covered the period 1992-2011. A second systematic database search identified preference-based value sets for generic multidimensional PROMs. The PROMs were categorised by conceptual basis (FDH status, QoL and HRQoL) and by target age (namely infants and pre-schoolers aged < 5 years, pre-adolescents aged 5-11, adolescents aged 12-18 and multi-age group coverage). Descriptive statistics assessed how PROM characteristics (domain coverage, respondent type and design) varied by conceptual basis and age categories. Involvement of children in PROM development and testing was assessed to understand content validity. Characteristics of value sets available for the childhood generic multidimensional PROMs were identified and compared., Results: We identified 89 PROMs, including 110 versions: 52 FDH, 29 QoL, 12 HRQoL, nine QoL-FDH and eight HRQoL-FDH measures; 20 targeted infants and pre-schoolers, 29 pre-adolescents, 24 adolescents and 37 for multiple age groups. Domain coverage demonstrated development trajectories from observable FDH aspects in infancy through to personal independence and relationships during adolescence. PROMs targeting younger children relied more on informant report, were shorter and had fewer ordinal scale points. One-third of PROMs were developed following qualitative research or surveys with children or parents for concept elicitation. There were 21 preference-based value sets developed by 19 studies of ten generic multidimensional childhood PROMs: seven were based on adolescents' stated preferences, seven were from adults from the perspective of or on behalf of the child, and seven were from adults adopting an adult's perspective. Diverse preference elicitation methods were used to elicit values. Practices with respect to anchoring values on the utility scale also varied considerably. The range and distribution of values reflect these differences, resulting in value sets with notably different properties., Conclusion: Identification and categorisation of generic multidimensional childhood PROMs and value sets by this review can aid the development, selection and interpretation of appropriate measures for clinical and population research and cost-effectiveness-based decision-making., (© 2022. The Author(s).)

Objective: Typically, migraine prevention trials focus on reducing migraine days. This narrow focus may not capture all that is important to people with migraine. Inconsistency in outcome selection across trials limits the potential for data pooling and evidence synthesis. In response, we describe the development of core outcome set for migraine (COSMIG)., Design: A two-stage approach sought to achieve international, multistakeholder consensus on both the core domain set and core measurement set. Following construction of a comprehensive list of outcomes, expert panellists (patients, healthcare professionals and researchers) completed a three-round electronic-Delphi study to support a reduction and prioritisation of core domains and outcomes. Participants in a consensus meeting finalised the core domains and methods of assessment. All stages were overseen by an international core team, including patient research partners., Results: There was a good representation of patients (episodic migraine (n=34) and chronic migraine (n=42)) and healthcare professionals (n=33) with high response and retention rates. The initial list of domains and outcomes was reduced from >50 to 7 core domains for consideration in the consensus meeting, during which a 2-domain core outcome set was agreed., Conclusion: International and multistakeholder consensus emerged to describe a two-domain core outcome set for reporting research on preventive interventions for chronic and episodic migraine: migraine-specific pain and migraine-specific quality of life. Intensity of migraine pain assessed with an 11-point Numerical Rating Scale and the frequency as the number of headache/migraine days over a specified time period. Migraine-specific quality of life assessed using the Migraine Functional Impact Questionnaire., Competing Interests: Competing interests: MU and RF are directors and shareholders of Clinvivo and are part of an academic partnership with Serco related to return to work initiatives. MU recused himself from any discussions related to the choice of Delphi platform for this study; is a chief investigator or co-investigator on multiple previous and current research grants from the UK National Institute for Health Research and Arthritis Research UK, and is a co-investigator on grants funded by the Australian NHMRC; is a National Institute for Health Research (NIHR) senior investigator; has received travel expenses for speaking at conferences from the professional organisations hosting the conferences; is a co-investigator on two NIHR-funded studies receiving additional support from Stryker; has accepted honoraria for teaching/lecturing from CARTA; and was an editor of the NIHR journal series and a member of the NIHR Journal Editors Group, for which he received a fee. MM serves on the advisory board for Abbott, Allergan, Eli Lilly, Medtronic, Novartis and TEVA; has received payment for the development of educational presentations from Allergan, electroCore, Eli Lilly, Medtronic, Novartis and TEVA; and has received research grants from Abbott, electroCore and Medtronic. SP is a director of Health Psychology Services, which, in part, provides psychological treatments for those with chronic pain., (© Author(s) (or their employer(s)) 2021. Re-use permitted under CC BY. Published by BMJ.)

Objectives: Consideration of health status in children and adolescents now includes broader concepts such as health-related quality-of-life (HRQoL). Globally, there is a need for relevant preference-based HRQoL measures (PBMs) for use in children and adolescents, yet measurement of HRQoL in these groups presents particular challenges. This article systematically reviews the available generic childhood PBMs and their application and cross-cultural validation in sub-Saharan African (sSA)., Methods: A systematic review of published literature from January 1, 1990, to February 8, 2017, was conducted using MEDLINE (through OvidSP), EMBASE (OvidSP), EconLit (EBSCOhost), PsycINFO, Web of Science, and PubMed., Results: A total of 220 full-text articles were included in a qualitative synthesis. Ten generic childhood PBMs were identified, of which 9 were adapted from adult versions and only 1 was developed specifically for children. None of the measures were originally developed in sSA or other resource-constrained settings. The Health Utilities Index Mark 3 (HUI3) and the EQ-5D-Y were the only measures that had been applied in sSA settings. Further, the HUI3 and the EQ-5D-Y were the only generic childhood PBM that attempted to establish cross-cultural validation in sSA. Five of the 6 of these validation studies were conducted using the EQ-5D-Y in a single country, South Africa., Conclusions: The findings show that application of generic childhood PBMs in sSA settings has hitherto been limited to the HUI3 and EQ-5D-Y. Most adaptations of existing measures take an absolutist approach, which assumes that measures can be used across cultures. Nevertheless, there is also need to ensure linguistic and conceptual equivalence and undertake validation across a range of sSA cultural contexts., (Copyright © 2021. Published by Elsevier Inc.)

Objectives: Assessments of health-related quality of life outcomes associated with preterm birth provide valuable complementary data to the objective biomedical assessments that have traditionally been reported. The objective of this study was to perform a systematic review and meta-analysis of health utility values associated with preterm birth generated using preference-based approaches to health-related quality of life measurement., Methods: Systematic searches of MEDLINE, Web of Science, EconLit, EMBASE, CINAHL, PsycINFO, the Cochrane Library and SCOPUS were performed, covering the literature from inception of the search engines to 26 June 2018. Studies reporting health utility values estimated using either direct or indirect utility elicitation methods and published in the English language were included. Central descriptive statistics and measures of variability surrounding health utility values for each study and control group, and differences between comparator groups, are reported for each included article. The effect of preterm birth on health utility values was estimated using a hierarchical linear model in a linear mixed-effects meta-regression., Results: Of 2139 unique articles retrieved, 20 articles met the inclusion criteria. All but one study used the Health Utilities Index (HUI) Mark 2 (HUI2) or Mark 3 (HUI3) measures as their primary health utility assessment method. All studies reporting health utility values for individuals born preterm or at low birthweight and a control group of individuals born at full term or normal birthweight reported lower utility values in the study groups, regardless of age at assessment, respondent type or valuation method. The meta-regression revealed that preterm birth was associated with a mean utility decrement of 0.066 (95% confidence interval [CI] 0.035-0.098; p < 0.001) after controlling for valuation method, respondent type, administration mode, year of publication, geographical region of study, study setting and age at assessment., Conclusion: Evidence identified by this review can act as data inputs into future economic evaluations of preventive or treatment interventions for preterm birth. Future research should focus particularly on estimating health utility values during the various stages of adulthood, and incorporating the effects of preterm birth on the preference-based health-related quality of life outcomes of parents and other family members.

Background: Overweight and obesity in children is associated with poor health-related quality of life (HRQoL), but the nuances of this relationship across different age and socio-demographic groups are not well-established. The aim of this study is to examine how the association between weight status and HRQoL changes with age and socioeconomic position (SEP) throughout childhood and adolescence., Methods: We used data from the Longitudinal Study of Australian Children (LSAC), a cohort study in which children were interviewed biennially from ages 4 to 17 years over seven waves of data. Measurements of HRQoL (using PedsQL TM ), body mass index (BMI), and socio-demographic characteristics were collected at each interview. Of the 4983 children recruited into the study, we included data from 4083 children (a total of 24,446 observations). We used generalised estimating equations to assess whether age and SEP modified the association between weight status and HRQoL, after controlling for sex, long-term medical condition, language spoken to child and maternal smoking status., Results: Age was a significant modifier of the association between weight status and HRQoL, with adjustment for known predictors of HRQoL (P < 0.001). At age 4, children with obesity had, on average, a 0.99 (95% CI 0.02-1.96) point lower PedsQL total score than children at healthy weight. This difference became clinically important by age 9 at 4.50 (95% CI 3.86-5.13) points and increased to 6.69 (95% CI 5.74-7.64) points by age 17. There was no evidence that SEP modified the relationship between weight status and HRQoL (P > 0.05)., Conclusions: Our results demonstrate that the relationship between overweight and obesity status and poor HRQoL is strengthened with increasing age through childhood and adolescence, but is not affected by SEP. Paediatricians, researchers and carers of children with obesity should acknowledge HRQoL outcomes, particularly for older children and adolescents.

Purpose: To systematically assess patterns and temporal changes in the measurement and valuation of childhood health utilities and associations between methodological factors., Methods: Studies reporting childhood health utilities using direct or indirect valuation methods, published by June 2017, were identified through PubMed, Embase, Web of Science, PsycINFO, EconLit, CINAHL, Cochrane Library and PEDE. The following were explored: patterns in tariff application; linear trends in numbers of studies/samples and paediatric cost-utility analyses (CUAs) and associations between them; changes in proportions of studies/samples within characteristic-based categories over pre-specified periods; impact of National Institute for Health and Care Excellence (NICE) guidance on primary UK research and associations between valuation method, age and methodological factors., Results: 335 studies with 3974 samples covering all ICD-10 chapters, 23 valuation methods, 12 respondent types and 42 countries were identified by systematic review. 34.0% of samples using indirect methods compatible with childhood applied childhood-derived tariffs. There was no association between numbers of studies/samples and numbers of CUAs. Compared to 1990-2008, 2009-June 2017 saw a significant fall in the proportion of studies using case series; significant compositional changes across ICD-10 chapters and significantly higher sample proportions using childhood-specific and adult-specific indirect valuation methods, and based on pre-adolescents, self-assessment, self-administration and experienced health states. NICE guidance was weakly effective in promoting reference methods. Associations between valuation method, age and methodological factors were significant., Conclusion: 1990-2017 witnessed significant changes in primary research on childhood health utilities. Health technology assessment agencies should note the equivocal effect of methodological guidance on primary research.

Purpose: Few studies focus on the health-related quality of life (HRQoL) of preschool children with overweight or obesity. This is relevant for evaluation of obesity prevention trials using a quality-adjusted life year (QALY) framework. This study examined the association between weight status in the preschool years and HRQoL at age 5 years, using a preference-based instrument., Methods: HRQoL [based on parent proxy version of the Health Utilities Index Mark 3 (HUI3)] and weight status were measured in children born in Australia between 2007 and 2009. Children's health status was scored across eight attributes of the HUI3-vision, hearing, speech, ambulation, dexterity, emotion, cognition and pain, and these were used to calculate a multi-attribute utility score. Ordinary least squares (OLS), Tobit and two-part regressions were used to model the association between weight status and multi-attribute utility., Results: Of the 368 children for whom weight status and HUI3 data were available, around 40% had overweight/obesity. After adjusting for child's sex, maternal education, marital status and household income, no significant association between weight status in the preschool years and multi-attribute utility scores at 5 years was found., Conclusions: Alternative approaches for capturing the effects of weight status in the preschool years on preference-based HRQoL outcomes should be tested. The application of the QALY framework to economic evaluations of obesity-related interventions in young children should also consider longitudinal effects over the life-course. Clinical Trial Registration The Healthy Beginnings Trial was registered with the Australian Clinical Trial Registry (ACTRNO12607000168459).

Importance: Open fractures of the lower limb occur when a broken bone penetrates the skin. There can be major complications from these fractures, which can be life-changing., Objectives: To assess the disability, rate of deep infection, and quality of life in patients with severe open fracture of the lower limb treated with negative pressure wound therapy (NPWT) vs standard wound management after the first surgical debridement of the wound., Design, Setting, and Participants: Multicenter randomized trial performed in the UK Major Trauma Network, recruiting 460 patients aged 16 years or older with a severe open fracture of the lower limb from July 2012 through December 2015. Final outcome data were collected through November 2016. Exclusions were presentation more than 72 hours after injury and inability to complete questionnaires., Interventions: NPWT (n = 226) in which an open-cell solid foam or gauze was placed over the surface of the wound and connected to a suction pump, creating a partial vacuum over the dressing, vs standard dressings not involving application of negative pressure (n = 234)., Main Outcomes and Measures: Disability Rating Index score (range, 0 [no disability] to 100 [completely disabled]) at 12 months was the primary outcome measure, with a minimal clinically important difference of 8 points. Secondary outcomes were complications including deep infection and quality of life (score ranged from 1 [best possible] to -0.59 [worst possible]; minimal clinically important difference, 0.08) collected at 3, 6, 9, and 12 months., Results: Among 460 patients who were randomized (mean age, 45.3 years; 74% men), 88% (374/427) of available study participants completed the trial. There were no statistically significant differences in the patients' Disability Rating Index score at 12 months (mean score, 45.5 in the NPWT group vs 42.4 in the standard dressing group; mean difference, -3.9 [95% CI, -8.9 to 1.2]; P = .13), in the number of deep surgical site infections (16 [7.1%] in the NPWT group vs 19 [8.1%] in the standard dressing group; difference, 1.0% [95% CI, -4.2% to 6.3%]; P = .64), or in quality of life between groups (difference in EuroQol 5-dimensions questionnaire, 0.02 [95% CI, -0.05 to 0.08]; Short Form-12 Physical Component Score, 0.5 [95% CI, -3.1 to 4.1] and Mental Health Component Score, -0.4 [95% CI, -2.2 to 1.4])., Conclusions and Relevance: Among patients with severe open fracture of the lower limb, use of NPWT compared with standard wound dressing did not improve self-rated disability at 12 months. The findings do not support this treatment for severe open fractures., Trial Registration: isrctn.org Identifier: ISRCTN33756652.

Background: A common feature of most reviews or catalogues of health utilities has been their focus on adult health states or derivation of values from adult populations. More generally, utility measurement in or on behalf of children has been constrained by several methodological concerns. The objective of this study was to conduct the first comprehensive systematic review and meta-analysis of primary utility data for childhood conditions and descriptors, and to determine the effects of methodological factors on childhood utilities., Methods: The review followed PRISMA guidelines. PubMed, Embase, Web of Science, PsycINFO, EconLit, CINAHL and Cochrane Library were searched for primary studies reporting health utilities for childhood conditions or descriptors using direct or indirect valuation methods. The Paediatric Economic Database Evaluation (PEDE) Porject was also searched for cost-utility analyses with primary utility values. Mean or median utilities for each of the main samples were catalogued, and weighted averages of utilities for each health condition were estimated, by valuation method. Mixed-effects meta-regression using hierarchical linear modeling was conducted for the most common valuation methods to estimate the utility decrement for each health condition category relative to general childhood population health, as well as the independent effects of methodological factors., Results: The literature searches resulted in 272 eligible studies. These yielded 3,414 utilities when all sub-groups were considered, covering all ICD-10 chapters relevant to childhood health, 19 valuation methods, 12 respondent types, 8 modes of administration, and data from 36 countries. A total of 1,191 utility values were obtained when only main study samples were considered, and these were catalogued by health condition or descriptor, and methodological characteristics. 1,073 mean utilities for main samples were used for fixed-effects meta-analysis by health condition and valuation method. Mixed-effects meta-regressions estimated that 53 of 76 ICD-10 delineated health conditions, valued using the HUI3, were associated with statistically significant utility decrements relative to general population health, whereas 38 of 57 valued using a visual analog scale (VAS) were associated with statistically significant VAS decrements. For both methods, parental proxy assessment was associated with overestimation of values, whereas adolescents reported lower values than children under 12 y. VAS responses were more heavily influenced by mode of administration than the HUI3., Conclusion: Utilities and their associated distributions, as well as the independent contributions of methodological factors, revealed by this systematic review and meta-analysis can inform future economic evaluations within the childhood context.

Introduction: Although HIV infection and its associated co-morbidities remain the commonest reason for hospitalisation in Africa, their impact on economic costs and health-related quality of life (HRQoL) are not well understood. This information is essential for decision-makers to make informed choices about how to best scale-up anti-retroviral treatment (ART) programmes. This study aimed to quantify the impact of HIV infection and ART on economic outcomes in a prospective cohort of hospitalised patients with high HIV prevalence., Methods: Sequential medical admissions to Queen Elizabeth Central Hospital, Malawi, between June-December 2014 were followed until discharge, with standardised classification of medical diagnosis and estimation of healthcare resources used. Primary costing studies estimated total health provider cost by medical diagnosis. Participants were interviewed to establish direct non-medical and indirect costs. Costs were adjusted to 2014 US$ and INT$. HRQoL was measured using the EuroQol EQ-5D. Multivariable analyses estimated predictors of economic outcomes., Results: Of 892 eligible participants, 80.4% (647/892) were recruited and medical notes found. In total, 447/647 (69.1%) participants were HIV-positive, 339/447 (75.8%) were on ART prior to admission, and 134/647 (20.7%) died in hospital. Mean duration of admission for HIV-positive participants not on ART and HIV-positive participants on ART was 15.0 days (95%CI: 12.0-18.0) and 12.2 days (95%CI: 10.8-13.7) respectively, compared to 10.8 days (95%CI: 8.8-12.8) for HIV-negative participants. Mean total provider cost per hospital admission was US$74.78 (bootstrap 95%CI: US$25.41-US$124.15) higher for HIV-positive than HIV-negative participants. Amongst HIV-positive participants, the mean total provider cost was US$106.87 (bootstrap 95%CI: US$25.09-US$106.87) lower for those on ART than for those not on ART. The mean total direct non-medical and indirect cost per hospital admission was US$87.84. EQ-5D utility scores were lower amongst HIV-positive participants, but not significantly different between those on and not on ART., Conclusions: HIV-related hospital care poses substantial financial burdens on health systems and patients; however, per-admission costs are substantially lower for those already initiated onto ART prior to admission. These potential cost savings could offset some of the additional resources needed to provide universal access to ART.

Introduction: Achilles tendon rupture affects over 11 000 people yearly in the UK, and the incidence is increasing. Controversy remains with regard to the best rehabilitation strategy for these patients. In operatively treated patients, functional bracing provides better outcomes compared with plaster casts. However, the role of functional bracing in non-operatively managed patients is unclear. This is the protocol for a multicentre randomised trial of plaster cast immobilisation versus functional bracing for patients with a non-operatively managed Achilles tendon rupture., Methods and Analysis: All adults presenting with a primary rupture of the Achilles tendon will be screened. Non-operatively treated patients will be eligible to take part in the trial. Broad eligibility criteria will ensure that the results of the study can be generalised to the wider patient population. Randomisation will be on a 1:1 basis. Both rehabilitation strategies are widely used within the National Health Service. Standardised protocols will be followed, and details of plaster material and brace will be as per the site's usual practice.A minimum of 330 patients will be randomised to obtain 90% power to detect a difference of 8 points in Achilles Tendon Total Rupture Score at 9 months. Quality of life and resource use will be collected at 3, 6 and 9 months. The differences between treatment groups will be assessed on an intention-to-treat basis. The results of the trial-based economic evaluation will be expressed in terms of incremental cost per quality-adjusted life-year gained., Ethics and Dissemination: The National Research Ethic Committee approved this study on 18 March 2016 (16/SC/0109).The National Institute for Health Research Health Technology Assessment monograph and a manuscript to a peer-reviewed journal will be submitted on completion of the trial (summer 2019). The results of this trial will substantially inform clinical practice on the clinical and cost-effectiveness of the treatment of this injury. This study has been registered on the International Standard Randomised Controlled Trial Number registry with reference no ISRCTN62639639., Competing Interests: Competing interests: MLC is a member of the UK NIHR HTA General Board. SEL is a member of the UK NIHR HTA Additional Capacity Funding Board, HTA end of life care and add-on studies, HTA Prioritisation Group and HTA Trauma Board., (© Article author(s) (or their employer(s) unless otherwise stated in the text of the article) 2017. All rights reserved. No commercial use is permitted unless otherwise expressly granted.)

Background: The scale-up of HIV self-testing (HIVST) in Africa is recommended, but little is known about how this novel approach influences economic outcomes following subsequent antiretroviral treatment (ART) compared with established facility-based HIV testing and counseling (HTC) approaches., Setting: HIV clinics in Blantyre, Malawi., Methods: Consecutive HIV-positive participants, diagnosed by HIVST or facility-based HTC as part of a community cluster-randomized trial (ISRCTN02004005), were followed from initial assessment for ART until 1-year postinitiation. Healthcare resource use was prospectively measured, and primary costing studies undertaken to estimate total health provider costs. Participants were interviewed to establish direct nonmedical and indirect costs over the first year of ART. Costs were adjusted to 2014 US$ and INT$. Health-related quality of life was measured using the EuroQol EQ-5D at each clinic visit. Multivariable analyses estimated predictors of economic outcomes., Results: Of 325 participants attending HIV clinics for assessment for ART, 265 were identified through facility-based HTC, and 60 through HIVST; 168/265 (69.2%) and 36/60 (60.0%), respectively, met national ART eligibility criteria and initiated treatment. The mean total health provider assessment costs for ART initiation were US$22.79 (SE: 0.56) and US$19.92 (SE: 0.77) for facility-based HTC and HIVST participants, respectively, and was US$2.87 (bootstrap 95% CI: US$1.01 to US$4.73) lower for the HIVST group. The mean total health provider costs for the first year of ART were US$168.65 (SE: 2.02) and US$164.66 (SE: 4.21) for facility-based HTC and HIVST participants, respectively, and comparable between the 2 groups (bootstrap 95% CI: -US$12.38 to US$4.39). EQ-5D utility scores immediately before and one year after ART initiation were comparable between the 2 groups. EQ-5D utility scores 1 year after ART initiation had increased by 0.129 (SE: 0.011) and 0.139 (SE: 0.027) for facility-based HTC and HIVST participants, respectively., Conclusions: Once HIV self-testers are linked into HIV services, their economic outcomes are comparable to those linking to services after facility-based HTC.

Background: Relatively little is known about the effects of mode of delivery on long-term health-related quality-of-life outcomes. Furthermore, no previous study has expressed these outcomes in preference-based (utility) metrics., Methods: The study population comprised 2,161 mothers recruited from a prospective population-based study in the East Midlands of England encompassing live births and stillbirths between 32 +0 and 36 +6 weeks' gestation and a sample of term-born controls. Perinatal data were extracted from the mothers' maternity records. Health-related quality-of-life outcomes were assessed at 12 months postpartum, using the EuroQol Five Dimensions (EQ-5D) measure with responses to the EQ-5D descriptive system converted into health utility scores. Descriptive statistics and multivariable analyses were used to estimate the relationship between the mode of delivery and health-related quality-of-life outcomes., Results: The overall health-related quality-of-life profile of the women in the study cohort mirrored that of the English adult population as revealed by national health surveys. A significantly higher proportion of women delivering by cesarean delivery reported some, moderate, severe, or extreme pain or discomfort at 12 months postpartum than women undergoing spontaneous vaginal delivery. Multivariable analyses, using the Ordinary Least Squares estimator revealed that, after controlling for maternal sociodemographic characteristics, cesarean delivery without maternal or fetal compromise was associated with a significant EQ-5D utility decrement in comparison to spontaneous vaginal delivery among all women (-0.026; p = 0.038) and among mothers of term-born infants (-0.062; p < 0.001). Among mothers of term-born infants, this result was replicated in models that controlled for all maternal and infant characteristics (utility decrement of -0.061; p < 0.001). The results were confirmed by sensitivity analyses that varied the categorization of the main exposure variable (mode of delivery) and the econometric strategy., Conclusions: Among mothers of term-born infants, cesarean delivery without maternal or fetal compromise is associated with poorer long-term health-related quality of life in comparison to spontaneous vaginal delivery. Further longitudinal studies are needed to understand the magnitude, trajectory, and underpinning mechanisms of health-related quality-of-life outcomes following different modes of delivery., (© 2016 Wiley Periodicals, Inc.)

Introduction: Mapping algorithms are increasingly being used to predict health-utility values based on responses or scores from non-preference-based measures, thereby informing economic evaluations., Objectives: We explored whether predictions in the EuroQol 5-dimension 3-level instrument (EQ-5D-3L) health-utility gains from mapping algorithms might differ if estimated using differenced versus raw scores, using the Roland-Morris Disability Questionnaire (RMQ), a widely used health status measure for low back pain, as an example., Methods: We estimated algorithms mapping within-person changes in RMQ scores to changes in EQ-5D-3L health utilities using data from two clinical trials with repeated observations. We also used logistic regression models to estimate response mapping algorithms from these data to predict within-person changes in responses to each EQ-5D-3L dimension from changes in RMQ scores. Predicted health-utility gains from these mappings were compared with predictions based on raw RMQ data., Results: Using differenced scores reduced the predicted health-utility gain from a unit decrease in RMQ score from 0.037 (standard error [SE] 0.001) to 0.020 (SE 0.002). Analysis of response mapping data suggests that the use of differenced data reduces the predicted impact of reducing RMQ scores across EQ-5D-3L dimensions and that patients can experience health-utility gains on the EQ-5D-3L 'usual activity' dimension independent from improvements captured by the RMQ., Conclusion: Mappings based on raw RMQ data overestimate the EQ-5D-3L health utility gains from interventions that reduce RMQ scores. Where possible, mapping algorithms should reflect within-person changes in health outcome and be estimated from datasets containing repeated observations if they are to be used to estimate incremental health-utility gains.

Background: Trial-based cost-utility analyses require health-related quality of life data that generate utility values in order to express health outcomes in terms of quality-adjusted life years (QALYs). Assessments of baseline health-related quality of life are problematic where trial participants are incapacitated or critically ill at the time of randomisation. This review aims to identify and critique methods for handling non-availability of baseline health-related quality of life data in trial-based cost-utility analyses within emergency and critical illness settings., Methods: A systematic literature review was conducted, following PRISMA guidelines, to identify trial-based cost-utility analyses of interventions within emergency and critical care settings. Databases searched included the National Institute for Health Research (NIHR) Journals Library (1991-July 2016), Cochrane Library (all years); National Health Service (NHS) Economic Evaluation Database (all years) and Ovid MEDLINE/Embase (without time restriction). Strategies employed to handle non-availability of baseline health-related quality of life data in final QALY estimations were identified and critiqued., Results: A total of 4224 published reports were screened, 19 of which met the study inclusion criteria (mean trial size 1670): 14 (74 %) from the UK, four (21%) from other European countries and one (5%) from India. Twelve studies (63%) were based in emergency departments and seven (37%) in intensive care units. Only one study was able to elicit patient-reported health-related quality of life at baseline. To overcome the lack of baseline data when estimating QALYs, eight studies (42%) assigned a fixed utility weight corresponding to either death, an unconscious health state or a country-specific norm to patients at baseline, four (21%) ignored baseline utilities, three (16%) applied values from another study, one (5%) generated utility values via retrospective recall and one (5%) elicited utilities from experts. A preliminary exploration of these methods shows that incremental QALY estimation is unlikely to be biased if balanced trial allocation is achieved and subsequent collection of health-related quality of life data occurs at the earliest possible opportunity following commencement of treatment, followed by an adequate number of follow-up assessments., Conclusion: Trial-based cost-utility analyses within emergency and critical illness settings have applied different methods for QALY estimation, employing disparate assumptions about the health-related quality of life of patients at baseline. Where baseline measurement is not practical, measurement at the earliest opportunity following commencement of treatment should minimise bias in QALY estimation.

Objectives: To compare assessments of health-related quality of life outcomes of care home residents reported by residents and care staff acting as proxies., Design: Linear regression and bivariate modelling of paired assessments from care home residents and care staff., Setting: 78 care homes in 2 regions in England., Participants: 556 care home residents aged 65 years or older and care staff., Main Outcome Measures: EQ-5D utility scores and responses to individual EQ-5D dimensions., Results: The depression status, cognitive function, physical function, activities of daily living, social engagement, pain and dementia diagnosis of care home residents all predicted discrepancies in EQ-5D reporting. For residents with no depressive symptoms, care staff underestimated residents' mean EQ-5D utility score by 0.134 (95% CI 0.097 to 0.171) and for those with severe depressive symptoms they overstated mean utility scores by 0.222 (95% CI 0.104 to 0.339). With increasing levels of pain in residents the care staff progressively estimated EQ-5D utilities above self-reported values; by 0.236 (95% CI 0.003 to 0.469) in those with the second highest pain scores. For those with no cognitive impairment, proxies overstated mean utility scores by 0.097 (95% CI 0.049 to 0.146), while for those with severe cognitive impairment they underestimated mean utility scores by 0.192 (95% CI 0.143 to 0.241)., Conclusions: Care home residents and staff appear to differ fundamentally in their assessment of the health-related quality of life, as measured by the EQ-5D, of residents with different levels of depression, pain and/or cognitive impairment. This could lead to interventions evaluated using proxy-based quality-adjusted life year estimates being wrongly rejected on cost-effectiveness grounds and may also make it difficult for carers to act as advocates with health and social care professionals for certain groups of residents. A more resident-focussed approach to assessment of health-related quality of life is needed., Competing Interests: Competing interests: None declared., (Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing/.)

Background: The aim of this study was to assess the psychometric properties, namely acceptability, validity, reliability, interpretability and responsiveness of the EuroQol EQ-5D (EQ-5D visual analogue (VAS) and EQ-5D (utility)), Short Form 12 Dimensions (SF-12), SF-6D and Michigan Hand Outcome Questionnaire (MHQ) in patients with rheumatoid arthritis (RA) of the hand., Methods: The empirical investigation was based upon data from a randomised controlled trial of 488 adults with rheumatoid arthritis who had pain and dysfunction of the hands and/or wrists. Participants completed the EQ-5D, SF-12 and MHQ at baseline and at 4 and 12 months follow up. Acceptability was measured using completion rates over time; construct validity using the "known groups" approach, based on pain troublesomeness; convergent validity using spearman's rho correlation (ρ); reliability using internal consistency (Cronbach's alpha); interpretability using minimal important differences (MID); and responsiveness using effect sizes and standardised response means (SRM) stratified by level of self-rated improvement in hand and wrist function or level of self-rated benefit and satisfaction from trial treatments., Results: At baseline, the study population had a mean age of 62.4 years, a mean MHQ score of 52.1 and included 76% women. The EQ-5D (utility) had the highest completion rates across time points. All instruments discriminated between pre-specified groups based on pain troublesomeness. Convergent validity analysis indicated that the MHQ score correlated strongly with the EQ-5D (ρ = 0.65) and SF-6D (ρ = 0.63) utility scores. The MHQ was most responsive at detecting change in indicators of RA pain severity between baseline and 4 months, whilst minimal important differences varied considerably across PROMs., Conclusions: The instruments evaluated in this study displayed varying psychometric properties in the context of RA of the hand. The selection of a preferred instrument in evaluative studies should ultimately depend on the relative importance placed on individual psychometric properties and the importance placed on generation of health utilities for economic evaluation purposes.

Study Design: This study examines the links between severity of whiplash associated disorder and costs and health outcomes., Objective: The study aims to estimate the economic costs and health-state utilities associated with disability levels and recovery trajectories after acute whiplash injury., Summary of Background Data: Data used were from the Managing Injuries of the Neck Trial, which collected information on 3851 people over a 12-month period after acute whiplash injury., Methods: Effects of whiplash associated disorder severity on economic costs (measured from a societal perspective and separately from a health and personal social services perspective) were estimated using two-part regression models, comprising probability of incurring a cost and the total cost, given one was incurred. Effects on health-state utilities (measured using the EQ-5D and SF-6D) were estimated using ordinary least squares regression, and two-part models as for costs., Results: There was a direct relationship between severity of disability after acute whiplash injury and economic costs. Between baseline and 4 months, average societal costs for those with no disability were £99.55 (UK£, 2009 prices), increasing to £668.53 for those with complete disability. Average societal costs for the whole sample were £234.15 over the first 4 months, decreasing to £127.51 between 8 and 12 months. Conversely, utility scores decreased with increased disability. The average EQ-5D utility score was 0.934 at 4 months for those with no disability, decreasing to 0.033 for those with complete disability. The average EQ-5D utility score for the whole sample increased from 0.587 immediately post-injury to 0.817 at 12 months. Relative costs and disutilities generated by the multivariate models are also presented by disability level and recovery trajectory., Conclusion: These results provide estimates of the costs and health-state utilities associated with disability levels and recovery trajectories after acute whiplash injury. They can be used to inform estimates of the cost-effectiveness of interventions targeting whiplash-associated disorders., Level of Evidence: 3.

Background: HIV self-testing (HIVST) has been found to be highly effective, but no cost analysis has been undertaken to guide the design of affordable and scalable implementation strategies., Methods: Consecutive HIV self-testers and facility-based testers were recruited from participants in a community cluster-randomised trial ( ISRCTN02004005 ) investigating the impact of offering HIVST in addition to facility-based HIV testing and counselling (HTC). Primary costing studies were undertaken of the HIVST service and of health facilities providing HTC to the trial population. Costs were adjusted to 2014 US$ and INT$. Recruited participants were asked about direct non-medical and indirect costs associated with accessing either modality of HIV testing, and additionally their health-related quality of life was measured using the EuroQol EQ-5D., Results: A total of 1,241 participants underwent either HIVST (n = 775) or facility-based HTC (n = 446). The mean societal cost per participant tested through HIVST (US$9.23; 95 % CI: US$9.14-US$9.32) was lower than through facility-based HTC (US$11.84; 95 % CI: US$10.81-12.86). Although the mean health provider cost per participant tested through HIVST (US$8.78) was comparable to facility-based HTC (range: US$7.53-US$10.57), the associated mean direct non-medical and indirect cost was lower (US$2.93; 95 % CI: US$1.90-US$3.96). The mean health provider cost per HIV positive participant identified through HIVST was higher (US$97.50) than for health facilities (range: US$25.18-US$76.14), as was the mean cost per HIV positive individual assessed for anti-retroviral treatment (ART) eligibility and the mean cost per HIV positive individual initiated onto ART. In comparison to the facility-testing group, the adjusted mean EQ-5D utility score was 0.046 (95 % CI: 0.022-0.070) higher in the HIVST group., Conclusions: HIVST reduces the economic burden on clients, but is a costlier strategy for the health provider aiming to identify HIV positive individuals for treatment. The provider cost of HIVST could be substantially lower under less restrictive distribution models, or if costs of oral fluid HIV test kits become comparable to finger-prick kits used in health facilities.

Background: The aim of this study was to assess the psychometric properties of the EQ-5D-3 L, the SF-12 v2 and its preference based derivative the SF-6D, and the St Georges Respiratory Questionnaire (SGRQ), in patients diagnosed with Acute Respiratory Distress Syndrome (ARDS)., Methods: Data from the Oscillation in ARDS (OSCAR) randomised unblinded clinical trial of 795 patients diagnosed with ARDS provided the foundation of this secondary psychometric analysis. The three source patient reported outcome measures (PROMs) (EQ-5D-3 L, SF-12 and SGRQ) were collected at both 6 and 12 months post randomisation. All measures were tested for acceptability, reliability, internal consistency, validity and responsiveness. Data from responders at 6 months was used to test for acceptability, reliability, known groups validity and internal responsiveness. Data from patients who responded at both 6 and 12 months was used to test for convergent validity and external responsiveness., Results: Rates of response at both 6 and 12 months post randomisation were 89.88 % for the EQ-5D-3 L, 77.38 % for the SF-6D, 71.43 % for both the physical and mental components of the SF-12 and 38.10 % for the SGRQ. All measures had a Cronbach's Alpha statistic higher than 0.7. For known group's validity, there was no difference in mean summary or utility scores between known groups for all PROMs with minimal effect sizes. All three source measures showed strong convergent and discriminant validity. There was consistent evidence that the SF-6D is an empirically valid and efficient alternative to the EQ-5D-3 L. The EQ-5D-3 L and SGRQ were more responsive compared to the SF-12 and SF-6D with the EQ-5D-3 L generating greater effect sizes than the SGRQ., Conclusion: The PROMs explored in this study displayed varying psychometric properties in the context of ARDS. Further research should focus on shortening the SGRQ whilst still maintaining its psychometric properties and mapping between the SGRQ and preference-based measures for future application within economic evaluations of respiratory focused interventions. The selection ofa preferred PROM for evaluative studies within the ARDS context should ultimately depend on the relative importance placed on individual psychometric properties and the importance placed on generation of health utilities for economic evaluation purposes.

Purpose: The Pediatric Quality of Life Inventory™ (PedsQL™) General Core Scales (GCS) were designed to provide a modular approach to measuring health-related quality of life in healthy children, as well as those with acute and chronic health conditions, across the broadest, empirically feasible, age groups (2-18 years). Currently, it is not possible to estimate health utilities based on the PedsQL™ GCS, either directly or indirectly. This paper assesses different mapping methods for estimating EQ-5D health utilities from PedsQL™ GCS responses., Methods: This study is based on data from a cross-sectional survey conducted in four secondary schools in England amongst children aged 11-15 years. We estimate models using both direct and response mapping approaches to predict EQ-5D health utilities and responses. The mean squared error (MSE) and mean absolute error (MAE) were used to assess the predictive accuracy of the models. The models were internally validated on an estimation dataset that included complete PedsQL™ GCS and EQ-5D scores for 559 respondents. Validation was also performed making use of separate data for 337 respondents., Results: Ordinary least squares (OLS) models that used the PedsQL™ GCS subscale scores, their squared terms and interactions (with and without age and gender) to predict EQ-5D health utilities had the best prediction accuracy. In the external validation sample, the OLS model with age and gender had a MSE (MAE) of 0.036 (0.115) compared with a MSE (MAE) of 0.036 (0.114) for the OLS model without age and gender. However, both models generated higher prediction errors for children in poorer health states (EQ-5D utility score <0.6). The response mapping models encountered some estimation problems because of insufficient data for some of the response levels., Conclusion: Our mapping algorithms provide an empirical basis for estimating health utilities in childhood when EQ-5D data are not available; they can be used to inform future economic evaluations of paediatric interventions. They are likely to be robust for populations comparable to our own (children aged 11-15 years in attendance at secondary school). The performance of these algorithms in childhood populations, which differ according to age or clinical characteristics to our own, remains to be evaluated.

Background: The aim of this study was to assess the acceptability, reliability, validity and responsiveness of the Short-Form Health Survey (SF-12) and its preference-based derivative (SF-6D), the EQ-5D and the Neck Disability Index (NDI) in patients recovering from acute whiplash injury., Methods: Data from the Managing Injuries of the Neck Trial of 3,851 patients with acute whiplash injury formed the basis of this empirical investigation. The EQ-5D and SF-12 were collected at baseline, and all three outcome measures were then collected at 4 months, 8 months and 12 months post-randomisation. The measures were assessed for their acceptability (response rates), internal consistency, validity (known groups validity and discriminant validity) and their internal and external responsiveness., Results: Response rates were broadly similar across the measures, with evidence of a floor effect for the NDI and a ceiling effect for the EQ-5D utility measure. All measures had Cronbach's α statistics of greater than 0.7, indicating acceptable internal consistency. The NDI and EQ-5D utility score correlated more strongly with the physical component scale of the SF-12 than the mental component scale, whilst this was reversed for the SF-6D utility score. The smaller standard deviations in SF-6D utility scores meant there were larger effect sizes for differences in utility score between patients with different injury severity at baseline than for the EQ-5D utility measure. However, the EQ-5D utility measure and NDI were both more responsive to longitudinal changes in health status than the SF-6D., Conclusions: There was no evidence of differences between the EQ-5D utility measure and NDI in terms of their construct validity, discriminant validity or responsiveness in patients with acute whiplash injury. However, both demonstrated superior responsiveness to longitudinal health changes than the SF-6D.

Objectives: To test whether health-related quality of life (HRQL) based on societal standards differs between very low birth weight/very preterm (VLBW/VP) and full-term (FT) adolescents using self and parent proxy reports. Also, to examine whether self and parent reported HRQL is explained by indicators of objective functioning in childhood., Study Design: This prospective cohort study followed 260 VLBW/VP adolescents, 12 VLBW/VP adolescents with disability, and 282 FT adolescents. Objective functioning was assessed at 8.5 years; HRQL was assessed at 13 years with the Health Utilities Index Mark 3 (HUI3)., Results: Adolescents reported more functional impairment than their parents especially in the psychological aspects of health. The mean difference in HUI3 multi-attribute utility scores between FT and VLBW/VP adolescents was small (parents: 0.91 [95% CI, 0.90, 0.92] vs 0.88 [95% CI, 0.86, 0.90]; adolescents: 0.87 [95% CI, 0.85, 0.89] vs 0.84 [95% CI, 0.82, 0.86]), but high for VLBW/VP adolescents with disabilities (0.18, 95% CI, -0.04, 0.40). Objective function did not predict HRQL in FT adolescents but contributed to prediction of HRQL in VLBW/VP adolescents without disabilities. Different indicators of objective functioning were important for adolescent vs parent reports. More variation in HUI3 scores was explained by objective function in VLBW/VP parent reports compared with adolescent reports (25% vs 18%)., Conclusions: VLBW/VP adolescents reported poorer HRQL than their FT peers in early adolescence. Improvement in HRQL as VLBW/VP children grow up is, at least partly, explained by exclusion of the most disabled in self reports by VLBW/VP adolescents and the use of different reference points by adolescents compared with parents., (Copyright © 2013 Mosby, Inc. All rights reserved.)

Background: Major behavioural risk factors, namely obesity, alcohol consumption, smoking, lack of fruit and vegetable intake and physical inactivity negatively impact on self-reported quality of life. However, little is known about their impact on preference-based measures of health-related quality of life commonly used to inform economic evaluations., Methods: Preference-based health-related quality of life outcomes associated with major behavioural risk factors were estimated using the EuroQol EQ-5D responses of 14 117 participants, aged ≥16 years, in the 2008 Health Survey for England. Multivariable linear regression was used to model the relationship between the five risk factors and EQ-5D utility scores. In addition, logistic regression was used to model their relationship to dichotomous reports of problems for each of the five EQ-5D dimensions., Results: Only one-third of participants had a body mass index within normal range, never drank alcohol, consumed at least five portions of fruit or vegetable/day or exercised regularly, while nearly half of participants were smokers or ex-smokers. In the fully adjusted multivariable analyses, reductions in EQ-5D utility scores (95% CI) of 0.105 (0.072 to 0.137), 0.062 (0.042 to 0.082) and 0.142 (0.129 to 0.155) were estimated for a body mass index ≥40 kg/m(2), heavy smoking (≥20 cigarettes/day) and physical inactivity, respectively. Hazardous alcohol consumption (men >4 and ≤8 units/day; women >3 and ≤6 units/day) and daily fruit and vegetable intake between three and less than five portions were associated with small positive effects on EQ-5D utility scores (p<0.05)., Conclusions: The high prevalence and substantial utility loss associated with obesity, smoking and physical inactivity highlight the potential impact that interventions aimed at their prevention or alleviation may have on population health.

Background: Childhood psychiatric disorders may have deleterious consequences through childhood and into adulthood., Aims: To estimate costs and preference-based health-related quality of life outcomes (health utilities) associated with a broad range of childhood psychiatric disorders during the eleventh year of life., Method: Participants in a whole-population study of extremely preterm children and term-born controls (EPICure) undertook psychiatric assessment using the Development and Well Being Assessment (DAWBA) and the Kaufman-Assessment Battery for Children. Questionnaires completed by parents and teachers described the children's utilisation of health, social and education services during the eleventh year of life. Parents also described their child's health status using the Health Utilities Index Mark 2 and Mark 3 health status classification systems. Descriptive and multiple regression techniques were used to explore the association between psychiatric disorders and economic outcomes., Results: The study presents detailed costs and health utilities associated with psychiatric disorders for the preterm population, term-born population and pooled study population, following appropriate controls., Conclusions: The results of this study should be used to inform future economic evaluations of interventions aimed at preventing childhood psychiatric disorders or alleviating their effects. Further research is required that identifies, measures and values the longer-term economic impacts of these disorders in a valid and reliable manner.

Objective: To estimate costs and health utilities associated with extremely preterm birth at approximately 11 years of age using evidence from a whole population study (the EPICure study)., Methods: The study population comprised surviving children born at 20 through 25 completed weeks of gestation in all 276 maternity units in the United Kingdom and Republic of Ireland from March through December 1995 and a control group of classmates born at full term, matched for age, sex, and ethnic group. Estimates of utilization of health, social, and education services were combined with unit costs derived from primary and secondary sources. Generalized liner regression was used to estimate the impact of extremely preterm birth on public sector costs during the 11th year of life. Suboptimal levels of function for each of the eight attributes of the Health Utilities Index Mark III (cognition, vision, hearing, speech, ambulation, dexterity, emotion, and pain) and multiplicative multi-attribute utility scores were compared between the extremely preterm children and their classmates. Tobit regressions were performed to explore the effects of gestational age at birth on the Health Utilities Index Mark III multiattribute utility score., Results: Mean (standard deviation [SD]) public sector costs over the 12-month period were 6484 UK pounds (5548 pounds) for the combined extremely preterm group and 4007 pounds (2537 pounds) for their classmates, generating a mean cost difference of 2477 UK pounds(bootstrap 95% confidence interval [CI] 1605 pounds, 3360 pounds) that was statistically significant (P < 0.001). The generalized linear models revealed that compared to birth at term, birth at < or =23 completed weeks, 24(+0)-24(+6) weeks and 25(+0)-25(+6) weeks gestation increased public sector costs by an average of 2417 UK pounds (95% CI 60 pounds, 4774 pounds; P = 0.044), 1528 UK pounds (95% CI 129 pounds, 2927 pounds; P = 0.032) and 1501 UK pounds (95% CI 428 pounds, 2574 pounds; P = 0.006), respectively. In all eight attributes of the Health Utilities Index Mark III, there were significantly higher proportions of suboptimal levels of function among the extremely preterm children (P < or = 0.05). The mean (SD) multiattribute utility score for the extremely preterm children as a cohort was 0.789 (0.264), compared to 0.956 (0.102) for the classmates born at term, a mean difference in utility score of 0.167 (95% CI 0.124, 0.209) that was statistically significant (P < 0.001). The Tobit regressions revealed that, compared to birth at term, birth at < or =23 completed weeks, 24(+0)-24(+6) weeks and 25(+0)-25(+6) weeks gestation reduced the Health Utilities Index Mark III multi-attribute utility score by an average of 0.312 (95% CI 0.169, 0.455; P < 0.001), 0.337 (95% CI 0.235, 0.439; P < 0.001) and 0.243 (95% CI 0.159, 0.327; P < 0.001), respectively., Conclusions: The results of this study should be used to inform the development of future economic evaluations of interventions aimed at preventing extremely preterm birth or alleviating its effects.

Background: Multi-attribute utility measures are preference-based health-related quality of life measures that have been developed to inform economic evaluations of health care interventions. The objective of this study was to compare the empirical validity of two multi-attribute utility measures (EQ-5D and SF-6D) based on hypothetical preferences in a large maternity population in England., Methods: Women who participated in a randomised controlled trial of additional postnatal support provided by trained community support workers represented the study population for this investigation. The women were asked to complete the EQ-5D descriptive system (which defines health-related quality of life in terms of five dimensions: mobility, self care, usual activities, pain/discomfort and anxiety/depression) and the SF-36 (which defines health-related quality of life, using 36 items, across eight dimensions: physical functioning, role limitations (physical), social functioning, bodily pain, general health, mental health, vitality and role limitations (emotional)) at six months postpartum. Their responses were converted into utility scores using the York A1 tariff set and the SF-6D utility algorithm, respectively. One-way analysis of variance was used to test the hypothetically-constructed preference rule that each set of utility scores differs significantly by self-reported health status (categorised as excellent, very good, good, fair or poor). The degree to which EQ-5D and SF-6D utility scores reflected alternative dichotomous configurations of self-reported health status and the Edinburgh Postnatal Depression Scale score was tested using the relative efficiency statistic and receiver operating characteristic (ROC) curves., Results: The mean utility score for the EQ-5D was 0.861 (95% CI: 0.844, 0.877), whilst the mean utility score for the SF-6D was 0.809 (95% CI: 0.796, 0.822), representing a mean difference in utility score of 0.052 (95% CI: 0.040, 0.064; p < 0.001). Both measures demonstrated statistically significant differences between subjects who described their health status as excellent, very good, good, fair or poor (p < 0.001), as well as monotonically decreasing utility scores (test for linear trend: p < 0.001). The SF-6D was between 29.1% and 423.6% more efficient than the EQ-5D at detecting differences in self-reported health status, and between 129.8% and 161.7% more efficient at detecting differences in the Edinburgh Postnatal Depression Scale score. In addition, the SF-6D generated higher area under the curve (AUC) scores generated by the ROC curves than the EQ-5D, indicating greater discriminatory power, although in all but one analysis the differences in AUC scores between the measures were not statistically significant., Conclusion: This study provides evidence that the SF-6D is an empirically valid and efficient alternative multi-attribute utility measure to the EQ-5D, and is capable of discriminating between external indicators of maternal health.

Objective: The objective of this study was to describe the health status and health-related quality of life preference-based outcomes of children with diagnosed bilateral permanent childhood hearing impairment and a comparison group of English-speaking children with normal hearing., Methods: We studied 120 children who were aged 7 to 9 years and had bilateral permanent childhood hearing impairment of moderate or greater severity, identified from a cohort of 156,733 children who were born in 8 districts of southern England, and 63 English-speaking children with normal hearing and the same place of birth and age at assessment. Principal caregivers were interviewed by using the Health Utilities Index Mark III questionnaire for proxy-assessed usual health status assessment. Levels of function within each of the 8 attributes of the Health Utilities Index Mark III (cognition, vision, hearing, speech, ambulation, dexterity, emotion, and pain) were recorded., Results: Bilateral permanent childhood hearing impairment is associated with significantly increased proportions of suboptimal levels of function and significantly lower single-attribute utility scores in 6 of the 8 attributes of the Health Utilities Index Mark III: vision, hearing, speech, ambulation, dexterity, and cognition. Compared with the children with normal hearing, the mean multiattribute utility score for the children with hearing impairment was significantly lower for both the whole group and the moderate, severe, and profound severity subgroups. The differences in the distributions of the multiattribute utility scores between the children with hearing impairment as a group and the children with normal hearing and between each of the severity subgroups and the children with normal hearing all were statistically significant., Conclusions: This study provides rigorous evidence of an association between bilateral permanent childhood hearing impairment and diminished health status and health-related quality of life preference-based outcomes during midchildhood.

Objective: The objective of this study was to describe the self-reported health status and health-related quality of life of British teenagers who are in mainstream schooling and were born before 29 weeks' gestational age compared with British teenagers who were born at term., Methods: All surviving children who were born at <29 weeks' gestation in the former Northern Region of England in 1983 and in the former Oxford Region of England and in Scotland in 1984 were eligible. A comparison group of teenagers who were born at term were also recruited. Children's responses to the Health Utilities Index Mark III were compared., Results: A total of 218 of the original 535 children who were born in the 3 regions during the study period were alive at 15 to 16 years of age. A complete Health Utilities Index Mark III record was available for 140 children in mainstream schools and for 108 control subjects. In 7 of the 8 attributes (vision, hearing, speech, emotion, pain, ambulation, and dexterity), there were no statistically significant differences in any functional impairment between the comparator groups. However, the preterm group did report a higher level of functional impairment in the cognition attribute (40.7% vs 25.0%). Although there was no difference in the median Health Utilities Index Mark III utility score between the 2 groups (0.93), there was a broader range of utility scores for the preterm group (0.07-1.0 vs 0.45-1.0 for the control group)., Conclusions: Despite objective evidence that children and teenagers who were born preterm have poorer health on average than term-born control subjects, this is not reflected in their own ratings of their health status and health-related quality of life at 15 to 16 years of age. The reasons for these differences need to be further explored.

Background: An important consideration for studies that derive utility scores using multi-attribute utility measures is the psychometric integrity of the measurement instrument. Of particular importance is the requirement to establish the empirical validity of multi-attribute utility measures; that is, whether they generate utility scores that, in practice, reflect people's preferences. We compared the empirical validity of EQ-5D versus SF-6D utility scores based on hypothetical preferences in a large, representative sample of the English population., Methods: Adult participants in the 1996 Health Survey for England (n=16 443) formed the basis of the investigation. The subjects were asked to complete the EQ-5D and SF-36 measures. Their responses were converted into utility scores using the York A1 tariff set and the SF-6D utility algorithm, respectively. One-way analysis of variance was used to test the hypothetically constructed preference rule that each set of utility scores differs significantly by self-reported health status (categorised as very good, good, fair, bad or very bad). The degree to which EQ-5D and SF-6D utility scores reflect alternative configurations of self-reported health status; illness, disability or infirmity, and medication use was tested using the relative efficiency statistic and receiver operating characteristic (ROC) curves., Results: The mean utility score for the EQ-5D was 0.845 (95% CI: 0.842, 0.849), whilst the mean utility score for the SF-6D was 0.799 (95% CI: 0.797, 0.802), representing a mean difference in utility score of 0.046 (95% CI: 0.044, 0.049; p<0.001). Bland-Altman plots displayed considerable lack of agreement between the two measures, particularly at the lower end of the utility scale. Both measures demonstrated statistically significant differences between subjects who described their health status as very good, good, fair, bad or very bad (p<0.001), as well as monotonically decreasing utility scores (test for linear trend: p<0.001). The SF-6D was between 30.9 and 100.4% more efficient than the EQ-5D at detecting differences in self-reported health status, and between 10.4 and 45.6% more efficient at detecting differences in illness, disability or infirmity and medication use. The area under the curve scores generated by the ROC curves were significantly higher for the SF-6D at the 0.1% significance level when self-reported health status was dichotomised as very good versus good, fair, bad or very bad. However, the AUC scores did not reveal any significant differences in the discriminatory powers of the measures when alternative configurations of illness, disability or infirmity and medication use were examined., Conclusions: This study provides evidence that the SF-6D is an empirically valid and efficient alternative multi-attribute utility measure to the EQ-5D, and is capable of discriminating between external indicators of health status. However, health economists should also consider other psychometric properties, such as practicality and reliability, when selecting either measure for evaluative purposes.

This paper considers the background to the rational allocation of health resources at DHA level within the context of the new purchaser/provider regime. A popular approach to this problem has been that of cost utility analysis, using quality adjusted life years (QALYs) as the measure of the benefit derived from a health intervention. This paper seeks to offer the practising public health physician an appraisal of the practical value and pitfalls of a QALY-based approach. To this end, the theoretical background to QALYs and their practical use are described and some of the practical and ethical problems and pitfalls are discussed. It is concluded that the QALY concept provides a useful tool in aiding decision making in the allocation of health care resources, but that it is too early for a universal application of the approach.

Background: Unstable ankle fractures represent a substantial burden of disease, accounting for a mean hospital stay of nine days, a mean cost of £4,491 per patient and 20,000 operations per year. There is variation in UK practice around weight-bearing instructions after operatively managed ankle fracture. Early weight-bearing may reduce reliance on health services, time off work, and improve functional outcomes. However, concerns remain about the potential for complications such as implant failure. This is the protocol of a multicentre randomised non-inferiority clinical trial of weight-bearing following operatively treated ankle fracture.Methods: Adults aged 18 years and over who have been managed operatively for ankle fracture will be assessed for eligibility. Baseline function (Olerud and Molander Ankle Score [OMAS]), health-related quality of life (EQ-5D-5L), and complications will be collected after informed consent has been obtained. A randomisation sequence has been prepared by a trial statistician to allow for 1:1 allocation to receive either instruction to weight-bear as pain allows from the point of randomisation, two weeks after the time of surgery ('early weight-bearing' group) or to not weight-bear for a further four weeks ('delayed weight -bearing' group). All other treatment will be as per the guidance of the treating clinician. Participants will be asked about their weight-bearing status weekly until four weeks post-randomisation. At four weeks post-randomisation complications will be collected. At six weeks, four months, and 12 months post-randomisation, the OMAS, EQ-5D-5L, complications, physiotherapy input, and resource use will be collected. The primary outcome measure is ankle function (OMAS) at four months post-randomisation. A minimum of 436 participants will be recruited to obtain 80% power to detect a non-inferiority margin of -6 points on the OMAS 4 months post-randomisation. A within-trial health economic evaluation will be conducted to estimate the cost-effectiveness of the treatment options.Discussion: The results of this study will inform national guidance with regards to the most clinically and cost-effective strategy for weight-bearing after surgery for unstable ankle fractures.Trial Registration: ISRCTN12883981 , Registered 02 December 2019. [ABSTRACT FROM AUTHOR]