{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,29]],"date-time":"2026-08-29T07:18:53Z","timestamp":1787987933778,"version":"build-2784847793"},"reference-count":21,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2020,10,15]],"date-time":"2020-10-15T00:00:00Z","timestamp":1602720000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2020,10,15]],"date-time":"2020-10-15T00:00:00Z","timestamp":1602720000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2020,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>The Hemophilia Joint Health Score (HJHS) was developed to detect early changes in joint health in children and adolescents with haemophilia. The HJHS is considered by some to be too time consuming for clinical use and this may limit broad adoption.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>This study was a first step to develop a shorter and\/or more convenient version of the HJHS for the measurement of joint function in children and young adults with haemophilia, by combining real\u2010life data and expert opinion.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A cross\u2010sectional multicenter secondary analysis on pooled data of published studies using the HJHS (0\u2010124, optimum score 0) in persons with haemophilia A\/B aged 4\u201030 was performed. Least informative items, scoring options and\/or joints were identified. An expert group of 19 international multidisciplinary experts evaluated the results and voted on suggestions for adaptations in a structured meeting (consensus set at\u00a0\u2265\u00a080%).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Original data on 499 persons with haemophilia from 7 studies were evaluated. Median age was 15.0\u00a0years [range 4.0\u201029.9], 83.2% had severe haemophilia and 61.5% received prophylaxis. Median (IQR) HJHS total was 6.0 (1.0\u201017.0). The items 'duration swelling' and 'crepitus' were identified as clinically less informative and appointed as candidates for reduction.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Analysis of 499 children and young adults with haemophilia showed that the HJHS is able to discriminate between children and adults and different treatment regimens. Reduction of the items 'duration swelling' and 'crepitus' resulted in the HJHS\n                      <jats:sub>short<\/jats:sub>\n                      , which had the same discriminative ability. Additional steps are needed to achieve a substantially shorter HJHS assessment.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.14180","type":"journal-article","created":{"date-parts":[[2020,10,15]],"date-time":"2020-10-15T10:39:16Z","timestamp":1602758356000},"page":"1072-1080","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":26,"title":["Evaluating international Haemophilia Joint Health Score (HJHS) results combined with expert opinion: Options for a shorter HJHS"],"prefix":"10.1111","volume":"26","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2920-2258","authenticated-orcid":false,"given":"Isolde A.R.","family":"Kuijlaars","sequence":"first","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht University Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2606-5104","authenticated-orcid":false,"given":"Janjaap","family":"van der Net","sequence":"additional","affiliation":[{"name":"Center for Child Development, Exercise and Physical Literacy Children\u2019s Hospital of the University Medical Center Utrecht Utrecht University Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7813-9665","authenticated-orcid":false,"given":"Brian M.","family":"Feldman","sequence":"additional","affiliation":[{"name":"Child Health Evaluative Sciences Program Research Institute The Hospital for Sick Children Toronto Ontario Canada"},{"name":"Department of Pediatrics Faculty of Medicine and the Institute of Health Policy Management &amp; Evaluation Dalla Lana School of Public Health University of Toronto Toronto Ontario Canada"},{"name":"Division of Rheumatology The Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4427-5159","authenticated-orcid":false,"given":"Magnus","family":"Aspdahl","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Clinic of Coagulation Disorders Karolinska University Hospital Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7413-1906","authenticated-orcid":false,"given":"Melanie","family":"Bladen","sequence":"additional","affiliation":[{"name":"Haemophilia Center Great Ormond Street Hospital for Children NHS Foundation Trust London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wypke","family":"de Boer","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Amsterdam UMC University of Amsterdam Amsterdam Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4276-6982","authenticated-orcid":false,"given":"Rub\u00e9n","family":"Cuesta\u2010Barriuso","sequence":"additional","affiliation":[{"name":"Department of Physiotherapy European University of Madrid Madrid Spain"},{"name":"Royal Victoria Eugenia Foundation Madrid Spain"},{"name":"Fishemo CEE Spanish Federation of Hemophilia Madrid Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3961-0207","authenticated-orcid":false,"given":"Ruth E.D.","family":"Matlary","sequence":"additional","affiliation":[{"name":"Department of Haematology Oslo University Hospital Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sharon M.","family":"Funk","sequence":"additional","affiliation":[{"name":"Hemophilia and Thrombosis Center University of Colorado Anschutz Medical Campus Aurora CO USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pamela","family":"Hilliard","sequence":"additional","affiliation":[{"name":"Child Health Evaluative Sciences Program Research Institute The Hospital for Sick Children Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judy A.","family":"John","sequence":"additional","affiliation":[{"name":"Department of PMR Christian Medical College Vellore India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2391-2016","authenticated-orcid":false,"given":"Christine L.","family":"Kempton","sequence":"additional","affiliation":[{"name":"Hemophilia of Georgia Center for Bleeding &amp; Clotting Disorders of Emory Emory University School of Medicine Atlanta GA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Piet","family":"de Kleijn","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht University Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5215-1482","authenticated-orcid":false,"given":"Marilyn","family":"Manco\u2010Johnson","sequence":"additional","affiliation":[{"name":"Hemophilia and Thrombosis Center University of Colorado Anschutz Medical Campus Aurora CO USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pia","family":"Petrini","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Clinic of Coagulation Disorders Karolinska University Hospital Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7715-9982","authenticated-orcid":false,"given":"Pradeep","family":"Poonnoose","sequence":"additional","affiliation":[{"name":"Department of Orthopaedics Christian Medical College Vellore India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean","family":"St\u2010Louis","sequence":"additional","affiliation":[{"name":"CHU Sainte\u2010Justine Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2375-8602","authenticated-orcid":false,"given":"Sylvia","family":"Thomas","sequence":"additional","affiliation":[{"name":"Department of Radiology Clementino Fraga Filho University Hospital Federal University of Rio de Janeiro Rio de Janeiro Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1910-999X","authenticated-orcid":false,"given":"Merel A.","family":"Timmer","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht University Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3576-7116","authenticated-orcid":false,"given":"Sonata Saulyte","family":"Trakymiene","sequence":"additional","affiliation":[{"name":"Clinic of Children\u2019s diseases Faculty of Medicine Vilnius University Vilnius Lithuania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leo","family":"van Vlimmeren","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Paediatric Physical Therapy Radboud university medical center Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7126-6613","authenticated-orcid":false,"given":"Kathelijn","family":"Fischer","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht University Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,10,15]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02909.x"},{"issue":"1","key":"e_1_2_10_3_1","first-page":"1","article-title":"Choosing outcome assessment tools in haemophilia care and research: A multidisciplinary perspective","volume":"23","author":"Fischer K","year":"2016","journal-title":"Haemophilia"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2008.01750.x"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/acr.20353"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12197"},{"key":"e_1_2_10_7_1","unstructured":"MokkinkLB TerweeCB PatrickDL et al.COSMIN checklist manual 2012."},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12090"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2012-12-470898"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12755"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12326"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13230"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13377"},{"key":"e_1_2_10_14_1","first-page":"1","article-title":"Systematic evaluation of hemophilic arthropathy in Lithuania","volume":"7","author":"Saulyte\u2010Trakymiene S","year":"2019","journal-title":"J Med Sci"},{"key":"e_1_2_10_15_1","unstructured":"International Prophylaxis Study Group.Hemophilia Joint Health Score 2.1Instruction Manual n.d."},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1017\/CBO9780511996214"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12290"},{"key":"e_1_2_10_18_1","first-page":"405","article-title":"Developing a new scoring scheme for the Hemophilia Joint Health Score 2.1. 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