{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T00:51:07Z","timestamp":1787014267609,"version":"build-2736575974"},"reference-count":30,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2015,4,9]],"date-time":"2015-04-09T00:00:00Z","timestamp":1428537600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2015,5]]},"abstract":"\n \n Background:<\/jats:bold>\n <\/jats:title>\n \n Joint bleeds (\n JB<\/jats:styled-content>\n ) are reported in a minority of patients with von Willebrand disease (\n VWD<\/jats:styled-content>\n ) but may lead to structural joint damage. Prevalence, severity and impact of\n JB<\/jats:styled-content>\n in\n VWD<\/jats:styled-content>\n are largely unknown.\n <\/jats:p>\n <\/jats:sec>\n \n \n Objectives:<\/jats:bold>\n <\/jats:title>\n \n The aim of this study was to assess\n JB<\/jats:styled-content>\n prevalence, onset, treatment and impact on health\u2010related quality of life (\n HR<\/jats:styled-content>\n \u2010QoL) and joint integrity in moderate and severe\n VWD<\/jats:styled-content>\n .\n <\/jats:p>\n <\/jats:sec>\n \n \n Methods:<\/jats:bold>\n <\/jats:title>\n \n In the Willebrand in the Netherlands study 804 moderate and severe\n VWD<\/jats:styled-content>\n patients [von Willebrand factor (\n VWF<\/jats:styled-content>\n ) activity \u226430U\u00a0dL\n \u22121<\/jats:sup>\n ] completed a questionnaire on occurrence, sites and consequences of\n JB<\/jats:styled-content>\n . To analyse\n JB<\/jats:styled-content>\n number, onset, treatment and impact on joint integrity we additionally performed a patient\u2013control study on medical file data comparing patients with\n JB<\/jats:styled-content>\n to age, gender, factor VIII (FVIII)\u2010 and\n VWF<\/jats:styled-content>\n activity matched\n VWD<\/jats:styled-content>\n patients without\n JB<\/jats:styled-content>\n .\n <\/jats:p>\n <\/jats:sec>\n \n \n Results:<\/jats:bold>\n <\/jats:title>\n \n Of all\n VWD<\/jats:styled-content>\n patients 23% (184\/804) self\u2010reported\n JB<\/jats:styled-content>\n . These 184 patients reported joint damage more often (54% vs. 18%,\n P<\/jats:italic>\n \u00a0<\u00a00.001) and had lower\n HR<\/jats:styled-content>\n \u2010QoL (SF36,\n P<\/jats:italic>\n \u00a0<\u00a00.05) compared to\n VWD<\/jats:styled-content>\n patients not reporting\n JB<\/jats:styled-content>\n . Of 55 patients with available\n JB<\/jats:styled-content>\n data, 65% had the first\n JB<\/jats:styled-content>\n before age 16. These 55 patients used more clotting factor concentrate (\n CFC<\/jats:styled-content>\n ; median dose 43 vs. 0 IE FVIII\u00a0kg\n \u22121<\/jats:sup>\n \u00a0year\n \u22121<\/jats:sup>\n ,\n P<\/jats:italic>\n \u00a0<\u00a00.001), more often had X\u2010ray joint damage (44% vs. 11%,\n P<\/jats:italic>\n \u00a0=\u00a00.001] and chronic joint pain (44% vs. 18%,\n P<\/jats:italic>\n \u00a0=\u00a00.008) compared to 55 control\n VWD<\/jats:styled-content>\n patients without\n JB<\/jats:styled-content>\n .\n <\/jats:p>\n <\/jats:sec>\n \n \n Conclusion:<\/jats:bold>\n <\/jats:title>\n \n In conclusion, joint bleeds are reported by 23% of moderate and severe\n VWD<\/jats:styled-content>\n patients, mostly start in childhood, are associated with more\n CFC<\/jats:styled-content>\n use, joint pain, lower\n HR<\/jats:styled-content>\n \u2010QoL and significantly more radiological and self\u2010reported joint damage.\n <\/jats:p>\n <\/jats:sec>","DOI":"10.1111\/hae.12670","type":"journal-article","created":{"date-parts":[[2015,4,9]],"date-time":"2015-04-09T03:08:53Z","timestamp":1428548933000},"source":"Crossref","is-referenced-by-count":50,"title":["Joint bleeds in von Willebrand disease patients have significant impact on quality of life and joint integrity: a cross\u2010sectional study"],"prefix":"10.1111","volume":"21","author":[{"given":"K. P. M.","family":"van Galen","sequence":"first","affiliation":[{"name":"Department of Van Creveldkliniek University Medical Center Utrecht Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Y. V.","family":"Sanders","sequence":"additional","affiliation":[{"name":"Department of Haematology Erasmus University Medical Center Rotterdam Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"U.","family":"Vojinovic","sequence":"additional","affiliation":[{"name":"Department of Haematology Erasmus University Medical Center Rotterdam Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J.","family":"Eikenboom","sequence":"additional","affiliation":[{"name":"Department of Thrombosis and Haemostasis and Einthoven Laboratory for Experimental Vascular Medicine Leiden University Medical Center Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M. H.","family":"Cnossen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Haematology Erasmus University Medical Center\u2010Sophia Children's Hospital Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R. E. G.","family":"Schutgens","sequence":"additional","affiliation":[{"name":"Department of Van Creveldkliniek University Medical Center Utrecht Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. G.","family":"van der Bom","sequence":"additional","affiliation":[{"name":"Jon J van Rood Center for Clinical Transfusion Medicine, Sanquin Research Leiden The Netherlands"},{"name":"Department of Clinical Epidemiology Leiden University Medical Center Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.","family":"Fijnvandraat","sequence":"additional","affiliation":[{"name":"Department of Pediatric Haematology Academisch Medisch Centrum Emma Children's Hospital Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"B. A. P.","family":"Laros\u2010Van Gorkom","sequence":"additional","affiliation":[{"name":"Department of Haematology Radboud University Medical Center Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.","family":"Meijer","sequence":"additional","affiliation":[{"name":"Department of Haematology University Medical Center Groningen University of Groningen Groningen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"F. W. G.","family":"Leebeek","sequence":"additional","affiliation":[{"name":"Department of Haematology Erasmus University Medical Center Rotterdam Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E. P.","family":"Mauser\u2010Bunschoten","sequence":"additional","affiliation":[{"name":"Department of Van Creveldkliniek University Medical Center Utrecht Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"the WiN Study Group","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2015,4,9]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood.V69.2.454.454"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538-7836.2006.02146.x"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1160\/TH12-04-0244"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2004.00991.x"},{"key":"e_1_2_10_6_1","first-page":"1","article-title":"Willebrand's disease in Sweden","volume":"238","author":"Silwer J","year":"1973","journal-title":"Acta Paediatr Scand Suppl"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.blre.2012.09.002"},{"key":"e_1_2_10_8_1","first-page":"339","article-title":"Blood\u2010induced joint damage: longterm effects in\u00a0vitro and in\u00a0vivo","volume":"30","author":"Hooiveld M","year":"2003","journal-title":"J Rheumatol"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2011.02669.x"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538-7836.2010.03864.x"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0031-1281032"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.12194"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02909.x"},{"key":"e_1_2_10_14_1","first-page":"A6518","article-title":"Von Willebrand disease in the Netherlands: the WiN study","volume":"158","author":"Sanders YV","year":"2014","journal-title":"Ned Tijdschr Geneeskd"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2005.01065.x"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12119"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.3109\/17453677008991542"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02025.x"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2004.00903.x"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1351-8216.2003.00826.x"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2003.00871.x"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12175"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1177\/145749690209100410"},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12258"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa067659"},{"key":"e_1_2_10_26_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02916.x"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2005.01124.x"},{"key":"e_1_2_10_28_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.1998.440413.x"},{"key":"e_1_2_10_29_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.mbc.0000167659.23262.18"},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2008.01851.x"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01573.x"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fhae.12670","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.12670","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.12670","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.12670","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,24]],"date-time":"2023-08-24T08:32:50Z","timestamp":1692865970000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.12670"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,4,9]]},"references-count":30,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2015,5]]}},"alternative-id":["10.1111\/hae.12670"],"URL":"https:\/\/doi.org\/10.1111\/hae.12670","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,4,9]]}}}