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- W3110771581 abstract "Orphan drugs are typically expensive, and, many payers have adapted the willingness-to-pay (WTP) thresholds for rare-disease (RD) treatments. However, a key debate is whether priority-setting should be utilitarian or egalitarian—maximising health gains for the greatest number of people within budget constraints versus advocating equal access to healthcare, regardless of costs. Ultimately, priority-setting should reflect public preferences. To explore this issue, a systematic literature review (SLR) was conducted on societal views about funding of RD treatments. Searches were conducted in Embase and Medline to identify relevant literature. All records were dual-screened, and relevant data were extracted from empirical studies that met selection criteria. This evidence was then analysed through narrative synthesis. The SLR included 19 publications reporting nine unique trade-off studies, three discrete-choice experiments and three studies that ranked the importance of value criteria for reimbursement decisions. Trade-off studies generally found that when treatment costs for RD and common diseases (CD) were equal, most respondents expressed indifference or chose equal allocation of resources. As costs for RD treatments increased, most respondents chose to prioritise funding for CD, indicating a preference towards maximising the number of patients treated. Studies investigating the relative importance of attributes on funding preferences generally reported that disease severity, treatment benefits, and unmet need had a greater impact on preferences than did ‘rarity’ of disease. Evidence suggests only limited public support for prioritising the funding of RD treatments based on ‘rarity’ alone, especially when such interventions are costly. These findings imply that the use of conventional frameworks with adapted WTP thresholds due to ‘rarity’, might not adequately reflect societal preferences for funding of orphan drugs. Instead, holistic frameworks capturing attributes, which substantially impact societal preferences (such as disease severity and unmet need), should be used to demonstrate the value of orphan products for reimbursement decisions." @default.
- W3110771581 created "2020-12-21" @default.
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- W3110771581 date "2020-12-01" @default.
- W3110771581 modified "2023-09-26" @default.
- W3110771581 title "PRO67 Does Society Support The Prioritisation Of High-Cost Treatments For Rare Diseases? A Systematic Literature Review" @default.
- W3110771581 doi "https://doi.org/10.1016/j.jval.2020.08.1803" @default.
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