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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.

Measuring and valuing health-related quality of life (HRQOL) in children can be challenging but is an important component for providing decision makers with accurate information to fund new interventions, including medicines and vaccines for public subsidy. We review funding submissions of medicines made to the Pharmaceutical Benefits Advisory Committee contained in public summary documents to examine the use of child-specific HRQOL measures in decision making in Australia. A sample frame of medicines used by children was derived from four sources. Public summary documents relating to these medicines were searched in the Pharmaceutical Benefits Advisory Committee web resources for whether they related to children (aged under 18 years) and contained HRQOL information and/or cost-utility analyses. Data about the use of utilities in decision making were extracted and analysed. Of the 1889 public summary documents available, 62 public summary documents (29 medicines) contained information pertaining to children and utilities. Of these, four public summary documents included child-specific HRQOL measures, 16 included adult HRQOL measures, 11 included direct elicitation and the HRQOL source was not defined in 31 documents. Excluding documents using child-specific HRQOL measures, we considered that in 85% of medicines, decision making uncertainty might have been reduced by using child-specific HRQOL measures. Despite the growing literature on economic analysis in paediatric populations, the use of child-specific HRQOL measures in submissions to the Pharmaceutical Benefits Advisory Committee was minimal. Submissions involved inconsistent approaches, use of adult measures and weights, and substantial gaps in evidence. We recommend the consistent use of child-specific measures to improve the evidence base for decisions about medicines for children in Australia.

Objectives To develop a cancer-specific multi-attribute utility instrument derived from the Functional Assessment of Cancer Therapy - General (FACT-G) health-related quality of life (HRQL) questionnaire. Methods We derived a descriptive system based on a subset of the 27-item FACT-G. Item selection was informed by psychometric analyses of existing FACT-G data (n = 6912) and by patient input (n = 82). We then conducted an online valuation survey, with participants recruited via an Australian general population online panel. A discrete choice experiment (DCE) was used, with attributes being the HRQL dimensions of the descriptive system and survival duration, and 16 choice-pairs per participant. Utility decrements were estimated with conditional logit and mixed logit modeling. Results Eight HRQL dimensions were included in the descriptive system: pain, fatigue, nausea, sleep, work, social support, sadness, and future health worry; each with 5 levels. Of 1737 panel members who accessed the valuation survey, 1644 (95%) completed 1 or more DCE choice-pairs and were included in analyses. Utility decrements were generally monotonic; within each dimension, poorer HRQL levels generally had larger utility decrements. The largest utility decrements were for the highest levels of pain (-0.40) and nausea (-0.28). The worst health state had a utility of -0.54, considerably worse than dead. Conclusions A descriptive system and preference-based scoring approach were developed for the FACT-8D, a new cancer-specific multi-attribute utility instrument derived from the FACT-G. The Australian value set is the first of a series of country-specific value sets planned that can facilitate cost-utility analyses based on items from the FACT-G and related FACIT questionnaires containing FACT-G items.

BackgroundUnderstandings of health and wellbeing are culturally bound. Many Aboriginal and Torres Strait Islander people perceive wellbeing and quality of life (QOL) differently from the Western biomedical models of health underpinning existing QOL instruments. Any instrument to measure the wellbeing of Aboriginal and Torres Strait Islander people should be culturally appropriate and safe, include relevant dimensions, and be informed by their own values and preferences. Existing QOL instruments do not meet these standards. This study will generate a new preference-based wellbeing measure, WM2Adults, for Aboriginal and Torres Strait Islander adults, underpinned by their values and preferences.MethodsA mixed methods approach will be used; we will employ decolonising methodologies, privilege Aboriginal and Torres Strait Islander voices and perspectives, and adopt a strengths-based approach rather than a deficit lens. Yarning Circles will be conducted with Aboriginal and Torres Strait Islander people across Australia. A candidate item pool will be developed from these data, on which psychometric analysis and validity testing will be undertaken to develop a descriptive system. Following finalisation of the descriptive system, wellbeing states will be valued using a quantitative preference-based approach (best-worst scaling) with a diverse sample of Aboriginal and Torres Strait Islander adults (n = 1000). A multinomial (conditional) logit framework will be used to analyse responses and generate a scoring algorithm for the new preference-based WM2Adults measure.DiscussionThe new wellbeing measure will have wide applicability in assessing the effectiveness and cost-effectiveness of new programs and services for Aboriginal and Torres Strait Islander people. Results will be disseminated through journals, conferences and policy forums, and will be shared with Aboriginal and Torres Strait Islander communities, organisations and research participants.

Objective Childhood cancer can have short‐ and long‐term impacts on parents’ finances and employment. It is important to understand how families adjust to the financial and employment changes caused by childhood cancer, the ongoing impacts after treatment completion, and which families need more targeted support. Qualitative research is necessary to facilitate an in‐depth understanding of the employment and financial impacts on families and to capture parents’ complex and nuanced experiences and perspectives. Methods We interviewed 56 parents of childhood cancer survivors (M = 2.13 years after treatment completion; 89% mothers) using the vocational and financial impact section of the Psychosocial Adjustment to Illness Scale–Carer Interview Form. We analyzed interviews using content analysis. Results Parents reported multiple sources of financial toxicity including travel to and from the hospital and needing to reduce their working hours during their child's cancer treatment. Workplace flexibility was an important factor to protect against unwanted vocational changes. After treatment completion, families living in low socioeconomic areas commonly reported ongoing financial difficulties. Mothers, particularly those who were on maternity leave when their child was diagnosed with cancer, reported ongoing employment impacts including unemployment. Conclusions Clinical staff including social workers could more consistently assess families’ financial distress and refer to professional services who can offer guidance for financial decision‐making as standard care. Flexible workplace agreements appear important for parents of children with cancer. Our findings can assist organizations to understand that cancer‐related disruptions are likely to continue after treatment completion, and therefore should offer benefits to parents where possible.

© 2019 John Wiley & Sons Ltd Background: Behaviour and emotional problems are highly prevalent in children with autism spectrum disorder (ASD). In typically developing children, attachment quality acts as a risk/protective factor for behavioural outcomes and adjustment, warranting investigation in children with ASD. Method: We investigated the relationship between attachment and child behaviour and emotional problems in children with ASD and comorbid intellectual disability. Data were collected from parent–child dyads where children were diagnosed with ASD and ID (n = 28) or other developmental disabilities (n = 20). Results: Children with ASD had higher levels of behaviour and emotional problems and more attachment difficulties than children with other developmental disabilities. Poorer attachment quality contributed uniquely to the variance in child behaviour and emotional problems. Conclusions: Interventions targeting behaviour and emotional problems in children with ASD may benefit from an attachment model which addresses the child's difficulty in using caregivers as a coregulatory agent of emotions.

BACKGROUND:Children with intellectual disability (ID) frequently have significant educational, social and health care needs, resulting in caregivers often experiencing a wide range of negative effects. This paper aims to determine the impact of childhood ID on caregivers' health-related quality of life (HRQoL) across co-morbid diagnostic groups. The second aim of this study is to determine the risk factors associated with lower HRQoL in this population. METHODS:Caregivers of a child with ID aged between 2 and 12 years old completed an online survey to determine their HRQoL using the EQ-5D-5L measure. They were also asked demographic questions and about their dependent child's level of behavioural and emotional difficulties. RESULTS:Of the total sample of 634 caregivers, 604 caregivers completed all five questions of the EQ-5D-5L. The mean age of caregivers was 39.1 years and 91% were women. Caregivers spent on average 66.6 h per week caring for their child related to their child's disability. The mean EQ-5D-5L score of caregivers was 0.80 (95% confidence interval: 0.79, 0.82), which is below the estimated Australian population norms (mean utility score of 0.92) for the age-equivalent population. Caregivers of children with autism spectrum disorders reported the lowest HRQoL (0.77, 95% confidence interval: 0.74, 0.79) of the five included co-morbid diagnostic groups. Caregivers with a lower income, a perceived low level of social support and children with higher degree of behavioural and emotional problems were likely to have a statistically lower HRQoL. CONCLUSIONS:This is the first study to produce utility values for caregivers of children with ID. The utility values can be used to compare health states and can be used to inform comparative cost-effectiveness analyses. Demonstrating that caregivers of children with ID have reduced HRQoL and that this is associated with the degree of behavioural and emotional problems has important policy implications, highlighting the potential for policy interventions that target behavioural and emotional problems to improve outcomes for caregivers.

OBJECTIVES:Debriefing questions can assess if respondents understand discrete choice experiments (DCEs) and are answering in a way consistent with theories of decision making and utility maximization. Nevertheless, there is limited literature about how often debriefing questions are included or how the results are used in health economics. The aim of this study was to conduct a survey of the frequency, type, and analysis of debriefing questions in health DCEs. METHODS:We conducted an online survey of authors of published health DCEs, asking about their use of debriefing questions, including frequency, type, and analysis. We descriptively analyzed the sample characteristics and responses. Free-text questions were analyzed with qualitative thematic analysis. RESULTS:We received 70 responses (43% response rate), of which 50% reported using debriefing questions. They were most commonly designed to assess difficulty (91%), understanding (49%), and attribute nonattendance (31%) rather than learning effects (3%) or monotonicity (11%). On average, 37% of debriefing questions were analyzed (range, 0% to 69%), and the results were used