Joint distraction in the treatment of haemophilic ankle arthropathy: clinical and structural efficacy, a pilot study

In the genetic bleeding disorder haemophilia, the most common complication is
joint damage due to recurrent joint bleeds. Besides the knees and elbows,
ankles are the most affected joints. Nowadays, in youngsters with haemophilia,
the ankle joint is the most affected joint. Surgical procedures like
arthrodesis (a permanent joint fusion) are often necessary. However, due to the
fast progressive joint degeneration starting at an early age, most patients are
still young (<55 years) and therefore surgery is postponed as long as possible.
In severe ankle osteoarthritis (another form of joint degeneration) joint
distraction appeared a good surgical alternative to postpone an ankle
arthrodesis for many years. This treatment might also be effective for
haemophilic ankle arthropathy (HAA) with the advantage of preservation of the
original joint. Recently three cases from clinical practice evaluated in
retrospect 2, 3, and 4 years after treatment, demonstrated good clinical and
structural efficacy.

This study is designed to gather prospective data on a) the clinical
effectiveness, b) the tissue structure changes, and c) the short term costs of
ankle joint distraction (AJD) in 10 patients with HAA during 10 years
follow-up.

This study is a prospective interventional pilot study with 3 years follow-up.
Haemophilia patients with severe complaints of ankle arthropathy in the
talocrural joint, insufficiently responding to analgetics and conservative
treatment, and leading to functional limitations are included. Ankle joint
distraction during 10 weeks is performed. Changes in symptoms are evaluated at
6 and 12 months post-surgery and thereafter yearly up to 3 years, and at 6 and
10 years post-surgery.

All ten patients included will be treated by ankle joint distraction during ten
weeks. An external frame will generate 5 mm distraction of the joint.

There is a direct benefit for the patients because good clinical benefit is
expected based on 3 cases in haemophilia and experience in patients with ankle
osteoarthritis. Preservation of the original joint for several years, thus
delaying the indicated joint fusion, is expected. Haemophilia patients with
arthropathy visit the Van Creveldkliniek regularly. For this study, patients
will visit the outpatient clinic with a comparable frequency as after a joint
fusion. Because of the study at baseline (twice), 6 months, 1, 2, 3, 6 and 10
years several questionnaires have to be filled in and physical tests performed.
Moreover, three and ten years after surgery an MRI is made solely for study
related purposes. Additionally, 10ml of blood and 5 ml of urine are collected
at all time points.
There is a risk due to the treatment and due to study related activities.
However, this risks associated with participation is considered minimal, based
on the experience with the 3 cases. The distraction frame will be applied under
general anaesthesia and after infusion of clotting factor concentrate. The risk
of pin tract infection or bleeding does exist, but both can be treated
effectively with antibiotics or clotting factor infusion respectively. After 10
weeks, the distraction frame will be removed, requiring general anaesthesia and
a bolus injection with clotting factor. Moreover, there is a negligible risk
due to x-ray radiation, and a small risk related to blood sampling.
The final outcome of the study will contribute to further development and
positioning of AJD in the treatment of haemophilic ankle arthropathy, which may
be of benefit to patients in the future and enable them to participate in their
society optimally.