Miners, Alexander Howard; (2000) The economics of replacement therapy for individuals with bleeding disorders. Doctoral thesis (Ph.D.), University College London (United Kingdom).

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Abstract

Individuals with haemophilia are deficient in essential clotting factors resulting in an increased tendency to bleed. Repeated bleeding into joints may cause haemophilic arthritis (HA). However, there is considerable interest from providers of haemophilia care in treating some individuals on a prophylactic basis to prevent bleeds, and hence joint damage, from occurring in the first instance. Prophylaxis was first administered at the Katharine Dormandy Haemophilia Centre (KDHC) in the late 1970s to some individuals with severe haemophilia, although full-time regimes were not introduced until the early 1980s. Data from individuals with severe haemophilia who were registered for treatment at the KDHC showed that following prophylaxis, the median incidence of bleeding had decreased significantly from 23.5 bleeds (range 1–107) per year in 1980 to 14 bleeds (range 0–52) per year by 1995 (P<0.0001). This said, however, individuals with severe haemophilia still recorded lower levels of health-related quality-of-life (HR-QoL) than individuals with mild / moderate haemophilia or the general UK male population even after adjusting for differences in age. Thus, significant scope exists for HR-QoL to be improved further. Using a unit clotting factor cost of 32.5 p/iu, a cost-utility analysis (CUA) showed that it cost an additional £46,500 per quality-adjusted life-year (QALY) and £8,600 per QALY to treat individuals with severe haemophilia A / vWD and severe haemophilia B with primary prophylaxis instead of on-demand respectively. However, the results from the CUA were not robust and both incremental cost-effectiveness ratios were found to be highly sensitive to a number of parameters including the unit clotting factor cost, the time between maintenance clotting factor infusions and the decision to discount future QALYs. Thus, further research over longer time periods is required to provide more accurate estimates of cost-effectiveness.

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