{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,8]],"date-time":"2026-09-08T06:08:09Z","timestamp":1788847689389,"version":"build-2803163510"},"reference-count":33,"publisher":"Oxford University Press (OUP)","issue":"3","license":[{"start":{"date-parts":[[2026,8,4]],"date-time":"2026-08-04T00:00:00Z","timestamp":1785801600000},"content-version":"vor","delay-in-days":34,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Netherlands Organization for Scientific Research"},{"name":"NWA-ORC","award":["NWA.1160.18.038"],"award-info":[{"award-number":["NWA.1160.18.038"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,7,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Hospital-level comparisons of patient-centered outcomes play a central role in value-based healthcare. In hemophilia care, several standardized sets of patient-centered outcomes have been developed to foster the implementation of value-based healthcare; however, their suitability for hospital-level comparisons has not yet been assessed. Therefore, we evaluated the suitability of patient-centered outcomes for hospital-level comparisons in hemophilia care.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We analyzed data from 807 adults with hemophilia enrolled in the 2019 HiN-6 study in the Netherlands. Twelve patient-centered outcomes were assessed. The suitability of these outcomes for hospital comparisons was evaluated using two criteria: (i) sensitivity to case mix, evaluated by Spearman\u2019s rank correlation between case-mix-adjusted and case-mix-unadjusted hospital-level scores and (ii) reliability of between-hospital differences, determined by rankability defined as the proportion of between-hospital variation not attributable to chance. High correlations indicate a minimal case-mix influence, while a high rankability reflects a high degree of reliability.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Most patient-centered outcomes were relatively insensitive to case mix (\u03c1\u2009&amp;gt;\u20090.70). Reliability of hospital differences was generally low (rankability &amp;lt;0.50). Only the annual bleeding rate and annual joint bleeding rate demonstrated moderate reliability with a rankability of 0.55 and 0.58, respectively.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Most patient-centered outcomes assessed were not suitable for comparing hemophilia care at the hospital level because of low reliability. Among the outcomes evaluated, the annual bleeding rate and annual joint bleeding rate appeared relatively more suitable for such comparisons.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/intqhc\/mzag105","type":"journal-article","created":{"date-parts":[[2026,8,3]],"date-time":"2026-08-03T11:18:05Z","timestamp":1785755885000},"source":"Crossref","is-referenced-by-count":0,"title":["Assessing the real-world value of hemophilia care: how suitable are patient-centered outcomes for hospital-level comparisons?"],"prefix":"10.1093","volume":"38","author":[{"given":"Diaz M","family":"Prameyllawati","sequence":"first","affiliation":[{"name":"Department of Public Health, Erasmus MC, University Medical Center Rotterdam , Rotterdam, 3015 GD,","place":["The Netherlands"]},{"name":"Department of Epidemiology and Health Economics, Julius Center for Health Sciences and Primary Care, University Medical Center Utrecht , Utrecht, 3584 CX,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samantha C","family":"Gouw","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology, Amsterdam UMC location University of Amsterdam , Amsterdam, 1105 AZ,","place":["The Netherlands"]},{"name":"Department of Clinical Epidemiology, Leiden University Medical Center , Leiden, 2333 ZA,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hester F","family":"Lingsma","sequence":"additional","affiliation":[{"name":"Department of Public Health, Erasmus MC, University Medical Center Rotterdam , Rotterdam, 3015 GD,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marjon H","family":"Cnossen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology and Oncology, Erasmus MC Sophia Children\u2019s Hospital, University Medical Center Rotterdam , Rotterdam, 3015 GD,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Floor C J I","family":"Heubel-Moenen","sequence":"additional","affiliation":[{"name":"Department of Hematology, Maastricht University Medical Center and Hemophilia Treatment Center Nijmegen-Eindhoven-Maastricht , Maastricht, 6229 HX,","place":["The Netherlands"]},{"name":"Cardiovascular Research Institute Maastricht (CARIM), Maastricht University , Maastricht, 6211 LK,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank W G","family":"Leebeek","sequence":"additional","affiliation":[{"name":"Department of Hematology, Erasmus MC, University Medical Center Rotterdam , Rotterdam, 3015 GD,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nick","family":"van Es","sequence":"additional","affiliation":[{"name":"Department of Vascular Medicine, Amsterdam University Medical Center , Amsterdam, 1105 AZ,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johanna G","family":"van der Bom","sequence":"additional","affiliation":[{"name":"Department of Clinical Epidemiology, Leiden University Medical Center , Leiden, 2333 ZA,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Renske M T","family":"ten Ham","sequence":"additional","affiliation":[{"name":"Department of Epidemiology and Health Economics, Julius Center for Health Sciences and Primary Care, University Medical Center Utrecht , Utrecht, 3584 CX,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martijn A H","family":"Oude Voshaar","sequence":"additional","affiliation":[{"name":"Department of Public Health, Erasmus MC, University Medical Center Rotterdam , Rotterdam, 3015 GD,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2026,8,4]]},"reference":[{"key":"2026090801422194000_mzag105-B1","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1111\/hae.14046","article-title":"WFH guidelines for the management of hemophilia, 3rd edition","volume":"26 Suppl 6","author":"Srivastava","year":"2020","journal-title":"Haemophilia"},{"key":"2026090801422194000_mzag105-B2","doi-asserted-by":"publisher","first-page":"20406207221085202","DOI":"10.1177\/20406207221085202","article-title":"International consensus recommendations on the management of people with haemophilia B","volume":"13","author":"Hart","year":"2022","journal-title":"Ther Adv Hematol"},{"key":"2026090801422194000_mzag105-B3","doi-asserted-by":"publisher","first-page":"102308","DOI":"10.1016\/j.rpth.2023.102308","article-title":"The evolution of hemophilia therapeutics: an illustrated review","volume":"8","author":"Espanol","year":"2024","journal-title":"Res Pract Thromb Haemost"},{"key":"2026090801422194000_mzag105-B4","doi-asserted-by":"crossref","first-page":"923","DOI":"10.1182\/blood.2019003777","article-title":"Gene therapy for hemophilia: a review on clinical benefit, limitations, and remaining issues","volume":"138","author":"Leebeek","year":"2021","journal-title":"Blood"},{"key":"2026090801422194000_mzag105-B5","author":"Porter","year":"2006"},{"key":"2026090801422194000_mzag105-B6","doi-asserted-by":"publisher","first-page":"e12488","DOI":"10.1002\/rth2.12488","article-title":"Patient-relevant health outcomes for hemophilia care: development of an international standard outcomes set","volume":"5","author":"van Balen","year":"2021","journal-title":"Res Pract Thromb Haemost"},{"key":"2026090801422194000_mzag105-B7","doi-asserted-by":"publisher","first-page":"437","DOI":"10.1111\/hae.14943","article-title":"The value-based healthcare approach to haemophilia: development of outcome measures for the evaluation of care of people with haemophilia","volume":"30","author":"Cortesi","year":"2024","journal-title":"Haemophilia"},{"key":"2026090801422194000_mzag105-B8","doi-asserted-by":"crossref","first-page":"504","DOI":"10.1056\/NEJMp1511701","article-title":"Standardizing patient outcomes measurement","volume":"374","author":"Porter","year":"2016","journal-title":"N Engl J Med"},{"key":"2026090801422194000_mzag105-B9","doi-asserted-by":"publisher","first-page":"329","DOI":"10.1080\/17474086.2024.2365929","article-title":"Outcome measures in hemophilia: current and future perspectives","volume":"17","author":"Benemei","year":"2024","journal-title":"Expert Rev Hematol"},{"key":"2026090801422194000_mzag105-B10","volume-title":"Measuring Quality of Care Methods and Applications to Acute Neurological Diseases","author":"Lingsma","year":"2010"},{"key":"2026090801422194000_mzag105-B11","doi-asserted-by":"publisher","first-page":"2394","DOI":"10.1111\/jth.15424","article-title":"Health and treatment outcomes of patients with hemophilia in The Netherlands, 1972-2019","volume":"19","author":"Hassan","year":"2021","journal-title":"J Thromb Haemost"},{"key":"2026090801422194000_mzag105-B12","author":"R Core Team","year":"2023"},{"key":"2026090801422194000_mzag105-B13","doi-asserted-by":"publisher","first-page":"1191","DOI":"10.1016\/j.jval.2019.12.014","article-title":"Effect of case-mix and random variation on breast cancer care quality indicators and their rankability","volume":"23","author":"Vos","year":"2020","journal-title":"Value Health"},{"key":"2026090801422194000_mzag105-B14","doi-asserted-by":"crossref","DOI":"10.4135\/9781412952644","volume-title":"Encyclopedia of Measurement and Statistics","author":"Salkind","year":"2007"},{"key":"2026090801422194000_mzag105-B15","first-page":"69","article-title":"Statistics corner: a guide to appropriate use of correlation coefficient in medical research","volume":"24","author":"Mukaka","year":"2012","journal-title":"Malawi Med J"},{"key":"2026090801422194000_mzag105-B16","author":"McBride","year":"2005"},{"key":"2026090801422194000_mzag105-B17","doi-asserted-by":"publisher","first-page":"869","DOI":"10.1136\/bmjqs.2010.048058","article-title":"Random variation and rankability of hospitals using outcome indicators","volume":"20","author":"van Dishoeck","year":"2011","journal-title":"BMJ Qual Saf"},{"key":"2026090801422194000_mzag105-B18","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1093\/qjmed\/hcp169","article-title":"Comparing and ranking hospitals based on outcome: results from The Netherlands stroke survey","volume":"103","author":"Lingsma","year":"2010","journal-title":"QJM"},{"key":"2026090801422194000_mzag105-B19","doi-asserted-by":"crossref","first-page":"8943","DOI":"10.34172\/ijhpm.8943","article-title":"Comprehensive evaluation of quality indicators: analyzing the Dutch Breast Cancer Audit","volume":"14","author":"Verheul","year":"2025","journal-title":"Int J Health Policy Manag"},{"key":"2026090801422194000_mzag105-B20","doi-asserted-by":"publisher","first-page":"523","DOI":"10.1007\/s10549-023-07158-w","article-title":"European quality indicators developed by the European Commission Initiative on Breast Cancer: a first nationwide assessment for the Dutch setting","volume":"203","author":"Schreuder","year":"2024","journal-title":"Breast Cancer Res Treat"},{"key":"2026090801422194000_mzag105-B21","doi-asserted-by":"publisher","first-page":"f167","DOI":"10.1136\/bmj.f167","article-title":"Patient reported outcome measures could help transform healthcare","volume":"346","author":"Black","year":"2013","journal-title":"BMJ"},{"key":"2026090801422194000_mzag105-B22","doi-asserted-by":"publisher","first-page":"754","DOI":"10.1111\/1468-0009","article-title":"Integrating the use of patient reported outcomes for both clinical practice and performance measurement: Views of experts from 3 countries","volume":"92","author":"Van Der Wees","year":"2014","journal-title":"Milbank Q"},{"key":"2026090801422194000_mzag105-B23","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1186\/s12955-024-02312-4","article-title":"The use of patient-reported outcome measures to improve patient-related outcomes - a systematic review","volume":"22","author":"Bonsel","year":"2024","journal-title":"Health Qual Life Outcomes"},{"key":"2026090801422194000_mzag105-B24","doi-asserted-by":"publisher","first-page":"100007","DOI":"10.1016\/j.hcr.2024.100007","article-title":"Patient-reported outcome measures in adults with hemophilia: a scoping review","volume":"1","author":"Bai","year":"2025","journal-title":"Healthcare and Rehabilitation"},{"key":"2026090801422194000_mzag105-B25","doi-asserted-by":"publisher","first-page":"430","DOI":"10.1111\/hae.12731","article-title":"Assessments of outcome in haemophilia - what is the added value of QoL tools?","volume":"21","author":"van den Berg","year":"2015","journal-title":"Haemophilia"},{"key":"2026090801422194000_mzag105-B26","doi-asserted-by":"publisher","first-page":"e208-209","DOI":"10.1111\/hae.12922","article-title":"Assessments of outcome in haemophilia - a patient perspective","volume":"22","author":"O\u2019Mahony","year":"2016","journal-title":"Haemophilia"},{"key":"2026090801422194000_mzag105-B27","doi-asserted-by":"publisher","first-page":"960","DOI":"10.1097\/MLR.0000000000000425","article-title":"Variation in patient-reported outcomes across hospitals following surgery","volume":"53","author":"Waljee","year":"2015","journal-title":"Med Care"},{"key":"2026090801422194000_mzag105-B28","doi-asserted-by":"publisher","first-page":"804","DOI":"10.1177\/0272989X13482523","article-title":"Hospital variation in patient-reported outcomes at the level of EQ-5D dimensions: evidence from England","volume":"33","author":"Gutacker","year":"2013","journal-title":"Med Decis Making"},{"key":"2026090801422194000_mzag105-B29","doi-asserted-by":"publisher","first-page":"127","DOI":"10.1002\/sim.2331","article-title":"Dichotomizing continuous predictors in multiple regression: a bad idea","volume":"25","author":"Royston","year":"2006","journal-title":"Stat Med"},{"key":"2026090801422194000_mzag105-B30","first-page":"195","article-title":"Evidence of selection bias and non-response bias in patient satisfaction surveys","volume":"39","author":"Compton","year":"2019","journal-title":"Iowa Orthop J"},{"key":"2026090801422194000_mzag105-B31","doi-asserted-by":"publisher","first-page":"1185","DOI":"10.1002\/sim.1970","article-title":"Funnel plots for comparing institutional performance","volume":"24","author":"Spiegelhalter","year":"2005","journal-title":"Stat Med"},{"key":"2026090801422194000_mzag105-B32","doi-asserted-by":"publisher","first-page":"376","DOI":"10.1097\/SLA.0b013e31819a47b1","article-title":"Funnel plots and their emerging application in surgery","volume":"249","author":"Mayer","year":"2009","journal-title":"Ann Surg"},{"key":"2026090801422194000_mzag105-B33","doi-asserted-by":"publisher","first-page":"95","DOI":"10.1111\/nep.13761","article-title":"Funnel plots of patient-reported outcomes to evaluate health-care quality: Basic principles, pitfalls and considerations","volume":"26","author":"van der Willik","year":"2021","journal-title":"Nephrology (Carlton)"}],"container-title":["International Journal for Quality in Health Care"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/intqhc\/advance-article-pdf\/doi\/10.1093\/intqhc\/mzag105\/70478740\/mzag105.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/intqhc\/article-pdf\/38\/3\/mzag105\/70478740\/mzag105.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/intqhc\/article-pdf\/38\/3\/mzag105\/70478740\/mzag105.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,8]],"date-time":"2026-09-08T05:42:31Z","timestamp":1788846151000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/intqhc\/article\/doi\/10.1093\/intqhc\/mzag105\/8750953"}},"subtitle":[],"editor":[{"given":"David","family":"Greenfield","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2026,7]]},"references-count":33,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2026,7,8]]}},"URL":"https:\/\/doi.org\/10.1093\/intqhc\/mzag105","relation":{},"ISSN":["1353-4505","1464-3677"],"issn-type":[{"value":"1353-4505","type":"print"},{"value":"1464-3677","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026,7]]},"published":{"date-parts":[[2026,7]]},"article-number":"mzag105"}}