{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T03:19:01Z","timestamp":1787800741529,"version":"build-2784847793"},"reference-count":35,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T00:00:00Z","timestamp":1787702400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T00:00:00Z","timestamp":1787702400000},"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"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Even with modern prophylaxis options, people living with haemophilia have significantly lower quality of life and joint health outcomes than those without.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>Our goal was to determine if real\u2010world health\u2010system costs are higher for people living with haemophilia A compared to those without and determine how joint\u2010health state impacts this cost.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We utilized a population\u2010level, retrospective study design using health administrative data to determine the impact of sex and joint\u2010health states on cross\u2010sectional cost. Cases were identified using diagnostic codes. Joint health states were determined in 30\u2010day intervals by stepping forward and backward from joint replacement surgeries. Control groups were established through propensity score matching, aligning participants based on demographic, economic and health\u2010related variables. Direct 30\u2010day medical costs were generated in 2022 CAD. The impact of sex and disease health phases on direct healthcare system costs was analyzed through a two\u2010part regression model.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>2341 eligible individuals with haemophilia A were identified. 26% were female. In pre\u2010severe joint damage, mean 30\u2010day costs were higher for patients with haemophilia A\u2014$910 (standard deviation SD: $4069) for females and $724 (SD: $3577) for males, compared to $469 (\u00b1$2126) and $352 (\u00b1$2188) for matched controls, respectively. The regression model showed that current joint health state, number of other comorbidities, haemophilia status and sex were important factors within healthcare system cost.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The results of this study indicate that even with haemophilia treatment there remains an increased healthcare system cost for those with haemophilia A, regardless of sex.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70388","type":"journal-article","created":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T02:46:40Z","timestamp":1787798800000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Population\u2010Level Real\u2010World Healthcare Costs in Men and Women With Haemophilia A"],"prefix":"10.1111","author":[{"given":"Sam","family":"Hirniak","sequence":"first","affiliation":[{"name":"School of Pharmacy, Faculty of Science University of Waterloo  Kitchener Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samantha S. M.","family":"Drover","sequence":"additional","affiliation":[{"name":"ICES  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea N.","family":"Edginton","sequence":"additional","affiliation":[{"name":"School of Pharmacy, Faculty of Science University of Waterloo  Kitchener Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dagmar M.","family":"Hajducek","sequence":"additional","affiliation":[{"name":"School of Pharmacy, Faculty of Science University of Waterloo  Kitchener Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alfonso","family":"Iorio","sequence":"additional","affiliation":[{"name":"Department of Health Research Methods, Evidence, and Impact McMaster University  Hamilton Ontario Canada"},{"name":"Department of Medicine McMaster University  Hamilton Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mhd. Wasem","family":"Alsabbagh","sequence":"additional","affiliation":[{"name":"School of Pharmacy, Faculty of Science University of Waterloo  Kitchener Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William W. L.","family":"Wong","sequence":"additional","affiliation":[{"name":"School of Pharmacy, Faculty of Science University of Waterloo  Kitchener Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,8,26]]},"reference":[{"key":"e_1_2_14_2_1","unstructured":"\u201cWorld Federation of Hemophilia Report on the Annual Global Survey \u201d October 2025 2024."},{"key":"e_1_2_14_3_1","doi-asserted-by":"publisher","DOI":"10.7326\/M19-1208"},{"key":"e_1_2_14_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2025.02.031"},{"key":"e_1_2_14_5_1","article-title":"A Comparative Analysis of the Bleeding Profile and Quality of Life Among Women With Hemophilia Genotype Compared to Other Bleeding Disorders","volume":"16","author":"Radhakrishnan N.","year":"2024","journal-title":"Cureus"},{"key":"e_1_2_14_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.15090"},{"key":"e_1_2_14_7_1","volume-title":"Women and Girls With Hemophilia","year":"2023"},{"key":"e_1_2_14_8_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2023-185067"},{"key":"e_1_2_14_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13546"},{"key":"e_1_2_14_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14979"},{"key":"e_1_2_14_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2005.01082.x"},{"key":"e_1_2_14_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.blre.2023.101164"},{"key":"e_1_2_14_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2022.12.029"},{"key":"e_1_2_14_14_1","doi-asserted-by":"crossref","first-page":"143","DOI":"10.1186\/s13023-021-01774-9","article-title":"Clinical, Humanistic, and Economic Burden of Severe Hemophilia B in the United States: Results From the CHESS US and CHESS US+ Population Surveys","volume":"16","author":"Burke T.","year":"2021","journal-title":"Orphanet Journal of Rare Diseases"},{"key":"e_1_2_14_15_1","doi-asserted-by":"crossref","first-page":"337","DOI":"10.1017\/S0266462316000544","article-title":"Cost\u2010Utility Analysis of Primary Prophylaxis, Compared With On\u2010Demand Treatment, for Patients With Severe Hemophilia Type A in Colombia","volume":"32","author":"Mejia A. E.","year":"2016","journal-title":"International Journal of Technology Assessment in Health Care"},{"key":"e_1_2_14_16_1","doi-asserted-by":"publisher","DOI":"10.18553\/jmcp.2023.29.6.647"},{"key":"e_1_2_14_17_1","doi-asserted-by":"publisher","DOI":"10.18553\/jmcp.2021.27.3.316"},{"key":"e_1_2_14_18_1","doi-asserted-by":"crossref","first-page":"106","DOI":"10.1186\/s13023-017-0660-y","article-title":"The Cost of Severe Haemophilia in Europe: The CHESS Study","volume":"12","author":"O'Hara J.","year":"2017","journal-title":"Orphanet Journal of Rare Diseases"},{"key":"e_1_2_14_19_1","doi-asserted-by":"publisher","DOI":"10.1080\/17474086.2020.1716726"},{"key":"e_1_2_14_20_1","unstructured":"\u201cTable 18\u201010\u20100005\u201001 Consumer Price Index Annual Average Not Seasonally Adjusted \u201dStatistics Canada."},{"key":"e_1_2_14_21_1","unstructured":"W.Wodchis K.Bushmeneva M.Nikitovic andI.McKillop \u201cGuidelines of Person\u2010Level Costing Using Administrative Databases in Ontario \u201d Toronto:Health System Performance Research Network 2013."},{"key":"e_1_2_14_22_1","doi-asserted-by":"publisher","DOI":"10.1191\/0962280202sm291ra"},{"key":"e_1_2_14_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mad.2021.111546"},{"key":"e_1_2_14_24_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10903-016-0351-0"},{"key":"e_1_2_14_25_1","doi-asserted-by":"publisher","DOI":"10.1093\/pch\/18.9.465"},{"key":"e_1_2_14_26_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13758"},{"key":"e_1_2_14_27_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40273-025-01484-1"},{"key":"e_1_2_14_28_1","unstructured":"D. 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P.Pearson \u201cValoctocogene Roxaparvovec and Emicizumab for Hemophilia A without Inhibitors: Effectiveness and Value \u201d November 20 2020 2020."},{"key":"e_1_2_14_29_1","doi-asserted-by":"publisher","DOI":"10.1080\/13696998.2020.1721508"},{"key":"e_1_2_14_30_1","doi-asserted-by":"crossref","DOI":"10.1111\/hae.13019","article-title":"Modelling Lifelong Effects of Different Prophylactic Treatment Strategies for Severe Haemophilia A","volume":"22","author":"Fischer K.","year":"2016","journal-title":"Haemophilia"},{"key":"e_1_2_14_31_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14979"},{"key":"e_1_2_14_32_1","doi-asserted-by":"crossref","first-page":"941","DOI":"10.2147\/PPA.S202773","article-title":"Real\u2010World Utilities and Health\u2010Related Quality\u2010of\u2010Life Data in Hemophilia Patients in France and the United Kingdom","volume":"13","author":"Carroll L.","year":"2019","journal-title":"Patient Preference and Adherence"},{"key":"e_1_2_14_33_1","doi-asserted-by":"crossref","first-page":"84","DOI":"10.1186\/s12955-018-0908-9","article-title":"The Impact of Severe Haemophilia and the Presence of Target Joints on Health\u2010Related Quality\u2010of\u2010Life","volume":"16","author":"O'Hara J.","year":"2018","journal-title":"Health and Quality of Life Outcomes"},{"key":"e_1_2_14_34_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13377"},{"key":"e_1_2_14_35_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2016-02-683169"},{"key":"e_1_2_14_36_1","unstructured":"D. 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