{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,19]],"date-time":"2026-06-19T13:54:48Z","timestamp":1781877288471,"version":"3.54.5"},"reference-count":7,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2026,2,26]],"date-time":"2026-02-26T00:00:00Z","timestamp":1772064000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,2,26]],"date-time":"2026-02-26T00:00:00Z","timestamp":1772064000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100000005","name":"U.S. Department of Defense","doi-asserted-by":"publisher","award":["W81XWH\u201017\u20101\u20100429"],"award-info":[{"award-number":["W81XWH\u201017\u20101\u20100429"]}],"id":[{"id":"10.13039\/100000005","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003579","name":"University of Toronto","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003579","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Laryngoscope Investig Oto"],"published-print":{"date-parts":[[2026,4]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>To characterize a proposed clinical trial outcome measure of weekly cumulative bleeding duration from daily nosebleed diaries, termed PRO\u2010CB (patient\u2010reported outcome of cumulative bleeding). We also assessed cumulative weekly gushing bleeding duration (PRO\u2010CGB) and evaluated the accuracy of patient recall on Epistaxis Severity Score (ESS) items 1\u20133 compared with daily diary data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>In this prospective observational study at the Toronto HHT Centre (St. Michael's Hospital, Unity Health Toronto), 23 patients from two clinical trials were observed during three\u2010month run\u2010in periods. Participants recorded each nosebleed episode daily, noting duration and intensity (gushing vs. non\u2010gushing). Data were analyzed using linear mixed\u2010effects models, correlation tests, and partial autocorrelation analyses.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Diary compliance was 98.8%. Mean weekly PRO\u2010CB was 71.2\u2009min (SD 81.1); mean episode duration was 5.9\u2009min (SD 12.6). Daily and monthly means were 10.1\u2009min (SD 20.1) and 307.6\u2009min (SD 311.6), respectively. Gushing episodes lasted significantly longer than non\u2010gushing (mean difference: 5.81\u2009min; 95% CI: 4.53\u20137.09). Bleeding patterns varied widely across individuals. PRO\u2010CGB also showed inter\u2010patient variability. PRO\u2010CB and ESS total scores were not significantly correlated (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.18,\n                      <jats:italic>r<\/jats:italic>\n                      \u2009=\u20090.39). ESS items 1 and 2 correlated strongly with diary data (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.001); item 3 did not. Agreement was 83% for item 1% and 70% for items 2 and 3.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Daily nosebleed diaries are a feasible and accurate tool for tracking epistaxis severity in HHT. Despite inter\u2010patient variability, PRO\u2010CB offers a meaningful outcome measure for epistaxis clinical trials.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Level of Evidence<\/jats:title>\n                    <jats:p>2<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/lio2.70354","type":"journal-article","created":{"date-parts":[[2026,3,9]],"date-time":"2026-03-09T00:12:00Z","timestamp":1773015120000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Characterizing Self\u2010Reported Daily Diary Epistaxis Measures in Hereditary Hemorrhagic Telangiectasia"],"prefix":"10.1002","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-5891-2157","authenticated-orcid":false,"given":"C. M.","family":"Tarulli","sequence":"first","affiliation":[{"name":"Toronto HHT Centre, Division of Respirology, Department of Medicine St. Michael's Hospital  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"X.","family":"Ma","sequence":"additional","affiliation":[{"name":"Toronto HHT Centre, Division of Respirology, Department of Medicine St. Michael's Hospital  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A. M.","family":"Bayoumi","sequence":"additional","affiliation":[{"name":"MAP Centre for Urban Health Solutions Li Ka Shing Knowledge Institute, St. Michael's Hospital  Toronto Ontario Canada"},{"name":"Division of General Internal Medicine St. Michael's Hospital  Toronto Ontario Canada"},{"name":"Department of Medicine University of Toronto  Toronto Ontario Canada"},{"name":"Institute of Health Policy, Management and Evaluation, University of Toronto  Toronto Ontario Canada"},{"name":"ICES  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Patel","sequence":"additional","affiliation":[{"name":"Toronto HHT Centre, Division of Respirology, Department of Medicine St. Michael's Hospital  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N. T.","family":"Vozoris","sequence":"additional","affiliation":[{"name":"Toronto HHT Centre, Division of Respirology, Department of Medicine St. Michael's Hospital  Toronto Ontario Canada"},{"name":"Li Ka Shing Knowledge Institute, St. Michaels Hospital, University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. M.","family":"Lee","sequence":"additional","affiliation":[{"name":"Toronto HHT Centre, Division of Respirology, Department of Medicine St. Michael's Hospital  Toronto Ontario Canada"},{"name":"Li Ka Shing Knowledge Institute, St. Michaels Hospital, University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M. E.","family":"Faughnan","sequence":"additional","affiliation":[{"name":"Toronto HHT Centre, Division of Respirology, Department of Medicine St. Michael's Hospital  Toronto Ontario Canada"},{"name":"Department of Medicine University of Toronto  Toronto Ontario Canada"},{"name":"Li Ka Shing Knowledge Institute, St. Michaels Hospital, University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,2,26]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.20818"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/alr.21374"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2016.11724"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoto.2020.3040"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023\u2010022\u201002539\u20108"},{"key":"e_1_2_12_7_1","unstructured":"DREXEL University College of Medicine \u201cEpistaxis Severity Score (ESS) for Hereditary Hemorrhagic Telangiectasia \u201dhttps:\/\/apps.med.drexel.edu\/hht\u2010ess\/."},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1056\/nejmoa2312749"}],"container-title":["Laryngoscope Investigative Otolaryngology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/lio2.70354","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/lio2.70354","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/lio2.70354","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/lio2.70354","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,19]],"date-time":"2026-06-19T13:12:14Z","timestamp":1781874734000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/lio2.70354"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,2,26]]},"references-count":7,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026,4]]}},"alternative-id":["10.1002\/lio2.70354"],"URL":"https:\/\/doi.org\/10.1002\/lio2.70354","archive":["Portico"],"relation":{},"ISSN":["2378-8038","2378-8038"],"issn-type":[{"value":"2378-8038","type":"print"},{"value":"2378-8038","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,2,26]]},"assertion":[{"value":"2025-10-13","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-28","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-02-26","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70354"}}