Introduction

Hemophilic arthropathy (HA) causes pain and limited range of motion, leading to limitations in daily activities [1]. The incidence of HA is decreasing, in line with a reduction of joint bleeding episodes from 25 bleeds per year per patient in 1971 to 2 bleeds in 2019 [2]. However, HA is still highly prevalent among patients with bleeding disorders, and emerging evidence suggests that subclinical bleeding also contributes to the development of HA [[3], [4], [5]]. Currently, one-fifth of the adolescents in the Netherlands with factor VIII (FVIII) activity <5% already show abnormalities on X-rays in one or more joints, increasing to almost all patients in their 40s [6,7].

Treatment of HA aims to relieve the symptoms and maintain physical functioning, primarily through nonsurgical interventions like physiotherapy and pain management. When pain and functional limitations persist, total joint replacement (TJR) is considered. However, the timing and indication of surgery differ from those of the general population. HA is already highly prevalent at a relatively young age, involves multiple joints, and results in severe joint deformities, synovial changes, and neovascularization, which complicate surgery and rehabilitation. Therefore, both clinicians and patients must constantly weigh the long-term benefits of arthroplasty against the burden of revision surgery and the high potential for complications [8]. Specifically, prosthetic joint infections (PJIs), aseptic loosening of the joint, and postoperative bleeding are prevalent after TJR in people with bleeding disorders [8,9], as they are associated with (subclinical) bleeding [10,11]. Consequently, prosthesis survival rates (PSRs) were historically worse for people with bleeding disorders than for the general population. Several studies have reported these PSRs in people with bleeding disorders, with follow-up periods extending up to 15 years for total knee arthroplasty (TKA), but only up to 5 years for total hip arthroplasty (THA) [5,8,[12], [13], [14], [15], [16], [17], [18], [19], [20], [21], [22], [23], [24]]. For TKA, the 15-year PSR was 84% in a European cohort [8]; for THA, the 5-year PSR was 91.9% in an American cohort [22,23]. In comparison, in a study of the Dutch general population (all indications, 86.4% osteoarthritis), patients with TKA and THA had a PSR of respectively 92.8% and 93.7% after 15 years [25,26].

Developments in hemophilia care, such introduction of the prophylactic factor (1970) and nonfactor therapies (2017), preoperative embolization in TKA (2000), diminished HIV and hepatitis C infection rates (mid-1990s) together with improved peri- and postoperative management have led to reduced complication rates over the last 2 decades [14,27,28]. Recent studies report improved survival rates that are approaching those observed in the general population, with a PSR of 94.7% at 15 years for TKA and 95.2% at 10 years for THA [12,13,20,[29], [30], [31], [32], [33], [34]]. However, these findings are based on relatively small cohorts, and PSRs extending past 2 decades are still lacking [27]. Therefore, we aimed to determine 15- and 25-year PSRs and long-term complication rates after TKA or THA in patients with end-stage HA. Furthermore, we aimed to identify predictors for failure of the prosthesis. We hypothesized that developments as preoperative embolization and improved (nonfactor) prophylactic treatment are predictors of prolonged prothesis survival [8], as both decrease bleeding risk. In contrast, we hypothesized that having an inhibitor, HIV/hepatitis C infections, postoperative bleeding, and PJI are related to a higher complication rate [9,10] and thus shorter survival.