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• W3113325231 abstract "Hemophilia A is a rare X-linked congenital disorder that affect males, caused by a mutation of clotting factor genes resulting in a deficiency of factor VIII (FVIII). Current standard-of-care treatments for hemophilia A in Canada include “on-demand” treatment of bleeds as they occur, and prophylactic treatment involving regular intravenous infusions of FVIII. The subcutaneous therapy emicizumab was recently approved for the treatment of hemophilia A (with and without inhibitors). The efficacy and safety of emicizumab relative to on-demand and prophylactic FVIII therapy was demonstrated in the HAVEN3 trial and companion non-interventional study. The objective of this study was to estimate utility values associated with hemophilia A health and treatment states from a Canadian societal perspective, including preferences related to treatment efficacy as well as frequency/mode of administration. A vignette-based time trade-off (TTO) utilities elicitation was undertaken to compare population preferences for 6 hemophilia health states describing prophylactic subcutaneous treatment, prophylactic FVIII intravenous infusions, and on-demand treatment, with varying bleed rates and frequency of treatment administration. Health state definitions were informed by HAVEN3 results regarding bleed and prophylaxis frequency, and supplemented with qualitative interviews of adult hemophilia patients and caregivers of pediatric patients (n=10), and clinical expert review. TTO interviews were conducted with N=82 general population respondents from across Canada. Mean utilities [95% CI] were highest for subcutaneous prophylaxis (0.90 [0.87-0.93]), followed by FVIII prophylaxis (0.81 [0.78-0.85]), with on-demand having the lowest utility (0.70 [0.65-0.76]). In regression analysis, subcutaneous treatment health states were associated with a utility increment of +0.1112. Additional bleeds and more frequent infusions were associated with lower utility values (-0.0027 per bleed and -0.0003 per infusion, respectively). Subcutaneous prophylaxis is associated with higher utility values compared to prophylactic intravenous treatment and on-demand treatment, while increased bleeds and -infusions are associated with reduced utility." @default.
• W3113325231 created "2020-12-21" @default.
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• W3113325231 date "2020-12-01" @default.
• W3113325231 modified "2023-10-14" @default.
• W3113325231 title "PRO64 Preferences for Hemophilia Treatments in Canada: A General Population Preference-Based Utility Study" @default.
• W3113325231 doi "https://doi.org/10.1016/j.jval.2020.08.1800" @default.
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