Background: Hemophilic ankle arthropathy (HAA) is a major cause of morbidity in hemophilia patients. If conservative treatment fails, major surgical interventions like arthrodesis are indicated. An alternative treatment to postpone these interventions and maintain joint mobility is desired. In osteoarthritis, joint distraction turned out to be a good joint-preserving alternative. Retrospective evaluation of three patients with HAA treated with ankle joint distraction (AJD) showed evident clinical and structural improvements.

Aims: To gather prospective data on clinical efficacy and tissue structure changes of AJD in HAA.

Methods: This ongoing first prospective study included patients ≥18 and ≤55 years with severe complaints of HAA, insufficiently responding to conservative treatment. AJD was performed during ten weeks by use of an external frame applying 5mm distraction. Clinical efficacy (questionnaires and performance tests) and tissue structure changes (MRI) were evaluated. The study was approved by the UMCU Medical Ethical Review Board and informed consent was obtained.

Results: One-, two- and three-year follow-up data are available for eight, seven and five patients respectively. Pain, evaluated by the Ankle Osteoarthritis Scale (AOS), decreased significantly. Functional limitations, assessed by the Hemophilia Activities List (HAL) and performance tests, improved over time, clearly adding to improved daily functioning (figure 1). Notably, the full arc of mobility was retained. MRI demonstrated improved osteochondral changes, whereas soft tissue changes remained unchanged (figure 2). The main complication during treatment was pin tract infection (n=6), commonly seen with external frames and effectively treated with a short course of antibiotics. There were no adverse bleeding events.

Conclusions: This explorative study shows that AJD in HAA results in decreased pain, improved function and tissue repair for a prolonged time. This joint-preserving treatment may have a great impact, especially in the relatively young hemophilia population. Prolonged follow-up is needed to determine the duration of efficacy in HAA.

[Figure 1: AOS pain; 0=no pain, 100=worst pain imaginable. HAL; 0=poor functional status,100=best functional status. Median values with IQR. ]

[Figure 2: Structural changes on MRI (sagittal images at baseline and one and three year after AJD).]

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ISTH Congress Abstracts - https://abstracts.isth.org/abstract/promising-prospective-data-on-the-effect-of-ankle-joint-distraction-in-hemophilic-arthropathy/