{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T04:06:32Z","timestamp":1791173192173,"version":"4.1.0"},"reference-count":41,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2016,10,26]],"date-time":"2016-10-26T00:00:00Z","timestamp":1477440000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/100004326","name":"Bayer AG","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100004326","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2016,11]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>The aim of replacement therapy in haemophilia is to improve Health\u2010Related Quality of Life (HRQoL) by preventing bleeding and arthropathy. However, the association of arthropathy with HRQoL is unknown.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>To explore the association of haemophilic arthropathy with HRQoL.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A<jats:italic>post hoc<\/jats:italic>analysis on patients with severe\/moderate haemophilia with SF36 questionnaire (SF36) and X\u2010rays of ankles, knees and elbows made within 2.5\u2010years. The SF36 scores of \u2018physical functioning\u2019 (SF36\u2010PF, range 0\u2013100, optimum 100) and Utility (SF6D\u2010Utility, range 0\u20131, optimum 1) and radiological Pettersson scores (PS, range 0\u201378, optimum 0) were calculated. The association of PS with reduced SF6D\u2010Utility and SF36\u2010PF (&lt;age\u2010specific normal values) was analysed using ROC analyses and multivariable logistic regression.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Overall, 176 assessments were analysed: 130 from the Van Creveldkliniek and 46 from a French multicenter study. Most patients had severe haemophilia (89.9%), evaluated at 26.6 years, and with a range 15.7\u201365.8. Overall PS median (interquartile range) was 16 (7\u201334), SF6D\u2010Utility was 0.76 (0.64\u20130.86) and SF36\u2010PF was 85 (60\u201395).<\/jats:p><jats:p>Receiver operating curve analysis identified a threshold PS of 21 points for both SF6D\u2010Utility (AUC 0.65) and SF36\u2010PF (AUC 0.76). In patients with PS &gt; 21 points, the risk of reduced SF6D\u2010Utility was stable (OR 4.16; 95% CI: 2.03\u20138.51) but SF36\u2010PF continued to decrease: compared to lowest PS, OR for reduced SF36\u2010PF was 5.69 (95% CI: 1.62\u201320.06) for PS 22\u201339 and 25.15 (95% CI: 6.53\u201396.81) for PS 40\u201378.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Health\u2010Related Quality of Life only showed a significant deterioration in patients with a Pettersson score of &gt;21 points. This suggests that HRQoL is relatively insensitive to early joint changes.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.13120","type":"journal-article","created":{"date-parts":[[2016,10,27]],"date-time":"2016-10-27T01:06:41Z","timestamp":1477530401000},"page":"833-840","source":"Crossref","is-referenced-by-count":36,"title":["The association of haemophilic arthropathy with Health\u2010Related Quality of Life: a<i>post hoc<\/i>analysis"],"prefix":"10.1111","volume":"22","author":[{"given":"K.","family":"Fischer","sequence":"first","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P.","family":"de Kleijn","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.","family":"Negrier","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Nursing Science and Sports University Medical Center Utrecht Utrecht the Netherlands"},{"name":"Unit\u00e9 d'H\u00e9mostase Clinique Center R\u00e9gional de traitement de l'H\u00e9mophilie Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E. P.","family":"Mauser\u2010Bunschoten","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P. R.","family":"van der Valk","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K. P. M.","family":"van Galen","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Willemze","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R.","family":"Schutgens","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek University Medical Center Utrecht Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,10,26]]},"reference":[{"key":"e_1_2_8_2_1","first-page":"5","article-title":"Haemophilia in Sweden VII. Incidence, treatment and prophylaxis of arthropathy and other musculo\u2010skeletal manifestations of haemophilia A and B","volume":"77","author":"Ahlberg A","year":"1965","journal-title":"Acta Orthop Scand"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/14651858.CD003429.pub4"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2011.02723.x"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2012-12-470898"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12169"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01562.x"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12009"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.307.6908.859"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1351-8216.2003.00823.x"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/0168-8510(90)90421-9"},{"key":"e_1_2_8_12_1","volume-title":"The SF36 Health Survey, Manual Interpretation Guide 1993","author":"Ware JE","year":"1993"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0167-6296(01)00130-8"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.mbc.0000178830.39526.ff"},{"key":"e_1_2_8_15_1","first-page":"722","article-title":"Multivariate analysis of factors influencing quality of life and utility in patients with haemophilia","volume":"86","author":"Trippoli S","year":"2001","journal-title":"Haematologica"},{"key":"e_1_2_8_16_1","first-page":"e842","article-title":"A survey of the outcome of prophylaxis, on\u2010demand, or combined treatment in 20\u201035 year old men with severe haemophilia in four European countries","volume":"17","author":"O'Mahony B","year":"2011","journal-title":"Haemophilia"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.2165\/00019053-200220110-00005"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2003.00811.x"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2010.029868"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12121"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2002.00581.x"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2004-05-2008"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2000.00358.x"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2002.00695.x"},{"issue":"1","key":"e_1_2_8_25_1","first-page":"s330","article-title":"Outcome in moderate haemophilia","volume":"12","author":"Uijl I","year":"2014","journal-title":"Blood Transfus"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1981.tb05741.x"},{"key":"e_1_2_8_27_1","doi-asserted-by":"crossref","first-page":"147","DOI":"10.1177\/028418518903000206","article-title":"Reproducibility of the Pettersson scoring system\u2013an interobserver study","volume":"30","author":"Erlemann R","year":"1989","journal-title":"Acta Radiol"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1097\/00005650-199206000-00002"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1002\/hec.1823"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0895-4356(98)00097-3"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.1586\/14737167.2015.1001372"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2005.01065.x"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1351-8216.2001.00119.x"},{"key":"e_1_2_8_34_1","first-page":"846","article-title":"Hepatitis C and health\u2010related quality of life among patients with haemophilia","volume":"90","author":"Posthouwer D","year":"2005","journal-title":"Haematologica"},{"key":"e_1_2_8_35_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12341"},{"key":"e_1_2_8_36_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13019"},{"key":"e_1_2_8_37_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2005.01054.x"},{"key":"e_1_2_8_38_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-004-7713-0"},{"key":"e_1_2_8_39_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0277-9536(99)00045-3"},{"key":"e_1_2_8_40_1","doi-asserted-by":"crossref","DOI":"10.1093\/oso\/9780198529446.001.0001","volume-title":"Methods for the Economic Evaluation of Health Care Programmes","author":"Drummond MJ","year":"2005"},{"key":"e_1_2_8_41_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00330-015-4013-8"},{"key":"e_1_2_8_42_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12755"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fhae.13120","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fhae.13120","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.13120","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.13120","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.13120","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,20]],"date-time":"2024-06-20T06:07:10Z","timestamp":1718863630000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.13120"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,10,26]]},"references-count":41,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2016,11]]}},"alternative-id":["10.1111\/hae.13120"],"URL":"https:\/\/doi.org\/10.1111\/hae.13120","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,10,26]]}}}