{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,14]],"date-time":"2026-02-14T05:27:21Z","timestamp":1771046841876,"version":"3.50.1"},"reference-count":12,"publisher":"MDPI AG","issue":"21","license":[{"start":{"date-parts":[[2021,10,29]],"date-time":"2021-10-29T00:00:00Z","timestamp":1635465600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJERPH"],"abstract":"<jats:p>Ankle joint distraction (AJD) has been described to be a valuable joint-sparing alternative to arthrodesis or arthroplasty; however, clinical endpoints associated to this surgical intervention are lacking. The current case report describes clinical and biomechanical outcome measures of ankle joint distraction in a 14-year-old patient with severe haemophilia A. Because of persistent and incapacitating pain and the poor response to conservative and invasive treatment options, ankle joint distraction was performed in this 14-year-old patient using an external fixator encompassing two Ilizarov full rings in the tibia and a foot ring fixed to the foot by four K-wires. State-of-the-art medical imaging and non-invasive skin marker-based 3D multi-segment foot modelling were performed in a pre- and post-operative stage. From a structural viewpoint, this AJD was a success since it improved and stabilised the osteo-cartilaginous lesions of the ankle. Biomechanical outcome measures associated with the 18-month follow-up were found to be suboptimal, showing an early plantarflexion pattern at the ankle joint during midstance and a tendency towards increased power absorption at the midfoot with peak power absorption being almost two times higher when compared to boys of the same age. From a functional viewpoint, we observed a clear reduction in the patients\u2019 physical activities until one year after AJD. Despite these functional and structural improvements, recurrent painful phenomena, including the development of a complex regional pain syndrome (CRPS) and a stress fracture of the third metatarsal bone, were observed which are probably related with the development of recurrent subchondral oedema.<\/jats:p>","DOI":"10.3390\/ijerph182111405","type":"journal-article","created":{"date-parts":[[2021,11,1]],"date-time":"2021-11-01T22:21:34Z","timestamp":1635805294000},"page":"11405","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Clinical and Biomechanical Progression after Ankle Joint Distraction in a Young Adolescent Patient with Haemophilia"],"prefix":"10.3390","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7510-7844","authenticated-orcid":false,"given":"Nicolas","family":"Haelewijn","sequence":"first","affiliation":[{"name":"Musculoskeletal Rehabilitation Research Group, Department of Rehabilitation Sciences, KU Leuven, Spoorwegstraat 12, B-8200 Brugge, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3829-6850","authenticated-orcid":false,"given":"Sebastien","family":"Lobet","sequence":"additional","affiliation":[{"name":"Service D\u2019h\u00e9matologie, Cliniques Universitaires Saint-Luc, Avenue Hippocrate 10, B-1200 Bruxelles, Belgium"},{"name":"Neuromusculoskeletal Lab (NMSK), Institut de Recherche Exp\u00e9rimentale et Clinique, Universit\u00e9 Catholique de Louvain, Secteur des Sciences de la Sant\u00e9, Avenue Mounier 53, B-1200 Brussels, Belgium"},{"name":"Secteur de Kin\u00e9sith\u00e9rapie, Cliniques Universitaires Saint-Luc, Avenue Hippocrate 10, B-1200 Brussels, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7114-201X","authenticated-orcid":false,"given":"An","family":"Van Damme","sequence":"additional","affiliation":[{"name":"Service D\u2019h\u00e9matologie P\u00e9diatrique, Cliniques Universitaires Saint-Luc, Avenue Hippocrate 10, B-1200 Bruxelles, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pierre-Louis","family":"Docquier","sequence":"additional","affiliation":[{"name":"Service D\u2019orthop\u00e9die et de Traumatologie, Cliniques Universitaires Saint-Luc, Avenue Hippocrate 10, B-1200 Bruxelles, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maarten","family":"Eerdekens","sequence":"additional","affiliation":[{"name":"Musculoskeletal Rehabilitation Research Group, Department of Rehabilitation Sciences, KU Leuven, Spoorwegstraat 12, B-8200 Brugge, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6559-6237","authenticated-orcid":false,"given":"Kevin","family":"Deschamps","sequence":"additional","affiliation":[{"name":"Musculoskeletal Rehabilitation Research Group, Department of Rehabilitation Sciences, KU Leuven, Spoorwegstraat 12, B-8200 Brugge, Belgium"},{"name":"Haute Ecole Leonard De Vinci, Institut D\u2019Enseignement Sup\u00e9rieur Parnasse Deux-Alice, Division of Podiatry, Avenue E. Mounier 84, B-1200 Sint-Lambrechts-Woluwe, Belgium"},{"name":"Department of Podiatry, Artevelde University of Applied Sciences, Voetweg 66, B-9000 Ghent, Belgium"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2021,10,29]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"250","DOI":"10.1111\/hae.13202","article-title":"Biomechanical markers and theoretical concepts related to haemophilic ankle and subtalar joint arthropathy: Introducing the term \u2018haemophilic tarsal pan-arthropathy\u2019","volume":"23","author":"Lobet","year":"2017","journal-title":"Haemophilia"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1210","DOI":"10.1111\/j.1365-2516.2008.01876.x","article-title":"Changing patterns of bleeding in patients with severe haemophilia A","volume":"15","author":"Stephensen","year":"2009","journal-title":"Haemophilia"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"2038","DOI":"10.1182\/blood-2015-01-528414","article-title":"Optimal treatment strategies for hemophilia: Achievements and limitations of current prophylactic regimens","volume":"125","author":"Oldenburg","year":"2015","journal-title":"Blood"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"1895","DOI":"10.1111\/j.1538-7836.2010.03962.x","article-title":"Blood-induced joint disease: The pathophysiology of hemophilic arthropathy","volume":"8","author":"Valentino","year":"2010","journal-title":"J. Thromb. Haemost."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"1207","DOI":"10.1007\/s00264-011-1455-8","article-title":"Total ankle arthroplasty versus ankle arthrodesis. Comparison of sports, recreational activities and functional outcome","volume":"36","author":"Schuh","year":"2012","journal-title":"Int. Orthop."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"61","DOI":"10.1007\/s11751-018-0305-2","article-title":"Distraction arthroplasty compared to other cartilage preservation procedures in patients with post-traumatic arthritis: A systematic review","volume":"13","author":"Rivera","year":"2018","journal-title":"Strateg. Trauma Limb. Reconstr."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"810","DOI":"10.1111\/j.1365-2516.2012.02805.x","article-title":"Joint distraction results in clinical and structural improvement of haemophilic ankle arthropathy: A series of three cases","volume":"18","author":"Biesma","year":"2012","journal-title":"Haemophilia"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"582","DOI":"10.1016\/j.joca.2005.03.002","article-title":"Prolonged clinical benefit from joint distraction in the treatment of ankle osteoarthritis","volume":"13","author":"Ploegmakers","year":"2005","journal-title":"Osteoarthr. Cartil."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"247","DOI":"10.1007\/s10439-019-02344-8","article-title":"Clinical Applicability of an Existing Proportionality Scheme in Three-Segment Kinetic Foot Models","volume":"48","author":"Eerdekens","year":"2020","journal-title":"Ann. Biomed. Eng."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"168","DOI":"10.1016\/j.jbiomech.2017.07.017","article-title":"Estimation of foot joint kinetics in three and four segment foot models using an existing proportionality scheme: Application in paediatric barefoot walking","volume":"61","author":"Deschamps","year":"2017","journal-title":"J. Biomech."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"372","DOI":"10.1016\/j.gaitpost.2018.03.051","article-title":"A novel approach for the detection and exploration of joint coupling patterns in the lower limb kinetic chain","volume":"62","author":"Deschamps","year":"2018","journal-title":"Gait Posture"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1097\/CORR.0000000000001443","article-title":"Loss of Mechanical Ankle Function Is Not Compensated by the Distal Foot Joints in Patients with Ankle Osteoarthritis","volume":"479","author":"Eerdekens","year":"2021","journal-title":"Clin. Orthop. Relat. Res."}],"container-title":["International Journal of Environmental Research and Public Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1660-4601\/18\/21\/11405\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T07:23:01Z","timestamp":1760167381000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1660-4601\/18\/21\/11405"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,10,29]]},"references-count":12,"journal-issue":{"issue":"21","published-online":{"date-parts":[[2021,11]]}},"alternative-id":["ijerph182111405"],"URL":"https:\/\/doi.org\/10.3390\/ijerph182111405","relation":{},"ISSN":["1660-4601"],"issn-type":[{"value":"1660-4601","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,10,29]]}}}