ArticlePloS one2025
Lifetime economic burden of hemophilia using a nationwide real-world healthcare data.
Article in PloS one, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
backgroundPatients with hemophilia require lifelong treatment, but the real-world lifetime economic burden of hemophilia remains unclear. This study aims to estimate the lifetime economic burden of hemophilia using real-world data, accounting for cost variation by disease phase and over time.
methodsMale patients with hemophilia A (PwHa) or B (PwHb) recorded in South Korea's Health Insurance Review and Assessment (HIRA) database from 2007 to 2022 were included. Survival was estimated using the rolling extrapolation method. A phase-specific costing approach was applied, distinguishing three phases: before hemophilic arthropathy (BH), after hemophilic arthropathy (AH), and 1 year before death (BD). Transition probabilities from BH to AH were calculated based on incidence rates of hemophilic arthropathy. Phase-specific annual costs were modeled using generalized estimating equations (GEEs), and predicted costs were multiplied by phase-specific probabilities from birth to estimated life expectancy to derive lifetime costs.
resultsEstimated life expectancy for PwHa (n = 2,624) and PwHb (n = 664) in South Korea between 2007-2022 was 76.13 and 77.54 years, respectively. The incidence rate of hemophilic arthropathy was 0.090 cases/person-year [95% confidence interval, 0.084-0.097] for PwHa and 0.080 [0.070-0.092] for PwHb, yielding transition probabilities from BH to AH of 0.086 (PwHa) and 0.077 (PwHb). Mean annual costs were highest in the BD phase, followed by AH and BH phases: PwHa (BH: $11,331; AH: $27,271; BD: $27,985); PwHb (BH: $15,567; AH: $38,659; BD: $55,985). Compared to PwHa, PwHb incurred 1.37 and 1.42 times higher BH and AH costs, respectively (p < 0.05). Estimated lifetime cost was 1.76 times higher for PwHb ($22.25 million) than PwHa ($12.62 million) in hypothetical patients born in 2000.
conclusionsHemophilia imposes a substantial economic burden across the lifespan, with PwHb incurring higher lifetime costs than PwHa. These real-world estimates can support more informed resource allocation and planning for hemophilia care.
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