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  • Longworth, Louise  (3)
  • 1
    Online Resource
    Online Resource
    Elsevier BV ; 2022
    In:  Molecular Genetics and Metabolism Vol. 135, No. 2 ( 2022-02), p. S58-
    In: Molecular Genetics and Metabolism, Elsevier BV, Vol. 135, No. 2 ( 2022-02), p. S58-
    Type of Medium: Online Resource
    ISSN: 1096-7192
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2022
    detail.hit.zdb_id: 1471393-7
    SSG: 12
    Location Call Number Limitation Availability
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  • 2
    Online Resource
    Online Resource
    The Journal of Health Economics and Outcomes Research ; 2023
    In:  Journal of Health Economics and Outcomes Research ( 2023-04-10), p. 80-88
    In: Journal of Health Economics and Outcomes Research, The Journal of Health Economics and Outcomes Research, ( 2023-04-10), p. 80-88
    Abstract: Background: Health state utilities are measures of health-related quality of life that reflect the value placed on improvements in patients’ health status and are necessary for estimation of quality-adjusted life-years. Health state utility data on Fabry disease (FD) are limited. In this study we used vignette (scenario) construction and valuation to develop health state utilities. Objectives: The aim of this study was to use vignette construction and valuation to estimate health state utility values suitable for inclusion in economic models of FD treatments. Methods: Health state vignettes were developed from semistructured qualitative telephone interviews with patients with FD and informed by published literature and input from an expert. Each vignette was valued in an online survey by members of the United Kingdom (UK) general population using the composite time trade-off (TTO) method, which aims to determine the time the respondent would trade to live in full health compared with each impaired health state. Results: Eight adults (50% women) with FD from the UK were interviewed. They were recruited via various approaches, including patient organizations and social media. The interviewees’ responses, evidence from published literature, and input from a clinical expert informed the development of 6 health state vignettes (pain, moderate clinically evident FD [CEFD], severe CEFD, end-stage renal disease [ESRD] , stroke, and cardiovascular disease [CVD]) and 3 combined health states (severe CEFD + ESRD, severe CEFD + CVD, and severe CEFD + stroke). A vignette valuation survey was administered to 1222 participants from the UK general population who were members of an external surveying organization and agreed to participate in this study; 1175 surveys were successfully completed and included in the analysis. Responses to TTO questions were converted into utility values for each health state. Pain was the highest valued health state (0.465), and severe CEFD + ESRD was the lowest (0.033). Discussion: Overall, mean utility values declined as the severity of the vignettes increased, indicating that respondents were more willing to trade life-years to avoid a severe health state. Conclusions: Health state vignettes reflect the effects of FD on all major health-related quality-of-life domains and may help to support economic modeling for treatment of FD.
    Type of Medium: Online Resource
    ISSN: 2327-2236
    Language: Unknown
    Publisher: The Journal of Health Economics and Outcomes Research
    Publication Date: 2023
    detail.hit.zdb_id: 2746906-2
    Location Call Number Limitation Availability
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  • 3
    Online Resource
    Online Resource
    The Journal of Health Economics and Outcomes Research ; 2023
    In:  Journal of Health Economics and Outcomes Research Vol. 10, No. 1 ( 2023-4-10)
    In: Journal of Health Economics and Outcomes Research, The Journal of Health Economics and Outcomes Research, Vol. 10, No. 1 ( 2023-4-10)
    Abstract: Background: Health state utilities are measures of health-related quality of life that reflect the value placed on improvements in patients’ health status and are necessary for estimation of quality-adjusted life-years. Health state utility data on Fabry disease (FD) are limited. In this study we used vignette (scenario) construction and valuation to develop health state utilities. Objectives: The aim of this study was to use vignette construction and valuation to estimate health state utility values suitable for inclusion in economic models of FD treatments. Methods: Health state vignettes were developed from semistructured qualitative telephone interviews with patients with FD and informed by published literature and input from an expert. Each vignette was valued in an online survey by members of the United Kingdom (UK) general population using the composite time trade-off (TTO) method, which aims to determine the time the respondent would trade to live in full health compared with each impaired health state. Results: Eight adults (50% women) with FD from the UK were interviewed. They were recruited via various approaches, including patient organizations and social media. The interviewees’ responses, evidence from published literature, and input from a clinical expert informed the development of 6 health state vignettes (pain, moderate clinically evident FD [CEFD], severe CEFD, end-stage renal disease [ESRD] , stroke, and cardiovascular disease [CVD]) and 3 combined health states (severe CEFD + ESRD, severe CEFD + CVD, and severe CEFD + stroke). A vignette valuation survey was administered to 1222 participants from the UK general population who were members of an external surveying organization and agreed to participate in this study; 1175 surveys were successfully completed and included in the analysis. Responses to TTO questions were converted into utility values for each health state. Pain was the highest valued health state (0.465), and severe CEFD + ESRD was the lowest (0.033). Discussion: Overall, mean utility values declined as the severity of the vignettes increased, indicating that respondents were more willing to trade life-years to avoid a severe health state. Conclusions: Health state vignettes reflect the effects of FD on all major health-related quality-of-life domains and may help to support economic modeling for treatment of FD.
    Type of Medium: Online Resource
    ISSN: 2327-2236
    Language: English
    Publisher: The Journal of Health Economics and Outcomes Research
    Publication Date: 2023
    detail.hit.zdb_id: 2746906-2
    Location Call Number Limitation Availability
    BibTip Others were also interested in ...
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