The humanistic and economic burden of problem joints for children and adults with moderate or severe haemophilia A: Analysis of the CHESS population studies

IntroductionAdequate prophylactic treatment and physical activity improve joint health and clinical outcomes for people with haemophilia A (HA). However, non-clinical joint-related burden of moderate (MHA) and severe (SHA) HA has not been well characterised. AimTo quantify the joint health-related humanistic and economic burden of MHA and SHA in Europe. MethodsA retrospective analysis of the cross-sectional CHESS population studies using a patient-centric measure of joint health (problem joints, PJs: chronic joint pain and/or limited range of movement due to compromised joint integrity with or without persistent bleeding) was conducted. Descriptive statistics summarised health-related quality of life (HRQoL), work productivity/activity impairment and costs by number of PJs (0, 1 or >= 2) and HA severity. ResultsA total of 1171 patients were included from CHESS-II (n = 468) and CHESS-PAEDs (n = 703). In both studies, 41 and 59% of patients had MHA and SHA, respectively. Prevalence of >= 2 PJs was similar with MHA and SHA (CHESS-II: 23 and 26%; CHESS-PAEDs: 4 and 3%, respectively). HRQoL was worse with an increasing number of PJs (CHESS-II: .81 vs. .66 with 0 and >= 2 PJs, respectively, for MHA; .79 vs. .51 for SHA; CHESS-PAEDs: .64 vs. .26 and .72 vs. .14). Total costs increased with increasing PJs regardless of severity in CHESS-II (euro2923 vs. euro22,536 with 0 and >= 2 PJs, respectively, for MHA; euro11,022 vs. euro27,098 for SHA) and CHESS-PAEDs (euro6222 vs. euro11,043 for MHA; euro4457 vs. euro14,039 for SHA). ConclusionPresence of PJs was associated with a substantial humanistic and economic burden on patients with MHA or SHA across the lifespan.