EHA Library - The official digital education library of European Hematology Association (EHA)

FIRST PROSPECTIVE RESULTS OF JOINT DISTRACTION IN SEVERE HEMOPHILIC ANKLE ARTHROPATHY
Author(s): ,
LFD Van Vulpen
Affiliations:
Van Creveldkliniek,University Medical Center,Utrecht,Netherlands
,
H Ch Vogely
Affiliations:
Orthopaedic Surgery,University Medical Center,Utrecht,Netherlands
,
G Roosendaal
Affiliations:
Van Creveldkliniek,University Medical Center,Utrecht,Netherlands
,
P de Kleijn
Affiliations:
Van Creveldkliniek,University Medical Center,Utrecht,Netherlands
,
PM van Roermund
Affiliations:
Orthopaedic Surgery,University Medical Center,Utrecht,Netherlands
,
REG Schutgens
Affiliations:
Van Creveldkliniek,University Medical Center,Utrecht,Netherlands
,
SC Mastbergen
Affiliations:
Rheumatology & Clinical Immunology,University Medical Center,Utrecht,Netherlands
FPJG Lafeber
Affiliations:
Rheumatology & Clinical Immunology,University Medical Center,Utrecht,Netherlands
(Abstract release date: 05/19/16) EHA Library. van Vulpen L. 06/11/16; 135226; S470
Lize F. D. van Vulpen
Lize F. D. van Vulpen
Contributions
Abstract
Abstract: S470

Type: Oral Presentation

Presentation during EHA21: On Saturday, June 11, 2016 from 12:15 - 12:30

Location: Room H4

Background
The ankle joint is the most affected joint in youngsters with hemophilia. Joint distraction, an effective treatment in ankle osteoarthritis, has the advantage of preservation of the original joint without compromising subsequent conservative surgeries like arthrodesis, if still needed. In three cases evaluated in retrospect, good clinical and structural efficacy in hemophilic ankle arthropathy was demonstrated.

Aims
The aim of the current study is to prospectively investigate the clinical effectiveness of ankle joint distraction in hemophilia.

Methods
Hemophilia patients (n=10; ≥18 and <55 years) were eligible in case of severe complaints of arthropathy in the tibiotalar joint causing functional limitations, despite analgesics and conservative treatment. Ankle joint distraction using an Ilizarov external fixator was performed during 10 weeks. Clinical effectiveness was evaluated using standard questionnaires and physical examination. Functional tests, X-ray and MRI examination were performed at baseline and 1-year follow-up.

Results
At the moment, a 12 months follow-up is available in 4 patients, three severe hemophilia A patients and one severe hemophilia B patient. Age at time of surgery ranged from 21 to 33 years. During distraction, none of the patients experienced bleeding. Pin tract infection, commonly seen with external frame use, occurred in 3 patients, and was treated effectively with oral antibiotics.Pain (visual analogue scale) decreased from 67 (47-79)mm at inclusion to 27 (7-84)mm at 6 months and 15 (1-43)mm at 12 months follow-up. Functional limitations, measured by the Haemophilia Activities List and the Ankle Osteoarthritis Scale, improved in three patients at 6 months, and in all four patients at 12 months. Functional tests improved considerably in all patients at 1-year follow up (e.g. 6-minutes walking test increased from 497 (434-560) to 621 (560-688) meters). Range of motion of the ankle was slightly decreased after 6 months due to stiffness of the ankle, but regained at 12 months in all patients.MRI revealed a decrease in volume of subchondral cysts and bone edema in all patients, and slight improvement of the joint space width in two patients (see figure).

Conclusion
This first prospective study investigating the efficacy of joint distraction in haemophilic ankle arthropathy, showed clear clinical and structural improvement in all patients at 1 yr follow-up. Although preliminary, these data indicate that joint distraction may be a promising treatment postponing more rigorous surgery like ankle arthrodesis in those patients not benefitting from conservative therapy.



Session topic: Bleeding disorders

Keyword(s): Hemophilia, Pain, Therapy
Abstract: S470

Type: Oral Presentation

Presentation during EHA21: On Saturday, June 11, 2016 from 12:15 - 12:30

Location: Room H4

Background
The ankle joint is the most affected joint in youngsters with hemophilia. Joint distraction, an effective treatment in ankle osteoarthritis, has the advantage of preservation of the original joint without compromising subsequent conservative surgeries like arthrodesis, if still needed. In three cases evaluated in retrospect, good clinical and structural efficacy in hemophilic ankle arthropathy was demonstrated.

Aims
The aim of the current study is to prospectively investigate the clinical effectiveness of ankle joint distraction in hemophilia.

Methods
Hemophilia patients (n=10; ≥18 and <55 years) were eligible in case of severe complaints of arthropathy in the tibiotalar joint causing functional limitations, despite analgesics and conservative treatment. Ankle joint distraction using an Ilizarov external fixator was performed during 10 weeks. Clinical effectiveness was evaluated using standard questionnaires and physical examination. Functional tests, X-ray and MRI examination were performed at baseline and 1-year follow-up.

Results
At the moment, a 12 months follow-up is available in 4 patients, three severe hemophilia A patients and one severe hemophilia B patient. Age at time of surgery ranged from 21 to 33 years. During distraction, none of the patients experienced bleeding. Pin tract infection, commonly seen with external frame use, occurred in 3 patients, and was treated effectively with oral antibiotics.Pain (visual analogue scale) decreased from 67 (47-79)mm at inclusion to 27 (7-84)mm at 6 months and 15 (1-43)mm at 12 months follow-up. Functional limitations, measured by the Haemophilia Activities List and the Ankle Osteoarthritis Scale, improved in three patients at 6 months, and in all four patients at 12 months. Functional tests improved considerably in all patients at 1-year follow up (e.g. 6-minutes walking test increased from 497 (434-560) to 621 (560-688) meters). Range of motion of the ankle was slightly decreased after 6 months due to stiffness of the ankle, but regained at 12 months in all patients.MRI revealed a decrease in volume of subchondral cysts and bone edema in all patients, and slight improvement of the joint space width in two patients (see figure).

Conclusion
This first prospective study investigating the efficacy of joint distraction in haemophilic ankle arthropathy, showed clear clinical and structural improvement in all patients at 1 yr follow-up. Although preliminary, these data indicate that joint distraction may be a promising treatment postponing more rigorous surgery like ankle arthrodesis in those patients not benefitting from conservative therapy.



Session topic: Bleeding disorders

Keyword(s): Hemophilia, Pain, Therapy

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