{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T04:25:33Z","timestamp":1772166333689,"version":"3.50.1"},"reference-count":10,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"Pettit Block-term grant, Division of Respirology, University of Toronto","award":["Pettit Block-term grant, Division of Respirology, University of Toronto"],"award-info":[{"award-number":["Pettit Block-term grant, Division of Respirology, University of Toronto"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Orphanet J Rare Dis"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>There has been concern that individuals living with Hereditary Hemorrhagic Telangiectasia (HHT) could be at higher risk for poor outcomes if infected with SARS-CoV2, the virus that causes COVID-19 disease. As literature is lacking on outcomes on COVID-19 infection and vaccination in HHT, the objectives of this study were to determine and assess outcomes in HHT, as well as quantify vaccination rates and vaccination side effects in a large cohort of individuals with HHT.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Method<\/jats:title>\n                    <jats:p>Individuals previously recruited to OUR HHT Registry at St. Michael\u2019s Hospital, Toronto were contacted for participation in this study. Data were collected during annual assessment through a series of questionnaires asking specifically about HHT complications, treatments, and symptom management, along with COVID infection and vaccination data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>We attempted to contact all 262 subjects recruited to the registry. Of these, 215 (82.1%) responded at least once regarding COVID-19 related inquiries between April 2020 and August 2022, and these individuals formed our study sample. Forty-nine COVID-19 infections were reported in 47\/215 (21.9%) individuals. Among 47 patients with recorded COVID-19 infection, 2\/47 (4.3%) required urgent care and 7\/47 (14.9%) were hospitalized following infection. Of the 7 individuals who were hospitalized, 3 (42.9%) required new supplemental oxygen. Zero deaths were reported due to COVID-19 infection. COVID vaccination history was available in 147\/215 (68.4%). Of these, 135\/147 (91.8%) of individuals reported vaccination and side effects were mild.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>While our sample population is much like the general HHT population with regards to gender, HHT symptoms, and genetics, study limitations including survivor bias, lack of vaccine effectiveness assessment, and participant reported data should be acknowledged.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Our results suggest that HHT patients are not at higher risk of severe infection with COVID-19 compared to the general population. Vaccination rates are high with only mild side effects being observed.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13023-025-03561-2","type":"journal-article","created":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T06:13:28Z","timestamp":1740809608000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Health outcomes following COVID-19 infection and vaccination in hereditary hemorrhagic telangiectasia"],"prefix":"10.1186","volume":"20","author":[{"given":"Christopher M.","family":"Tarulli","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiayi","family":"Ma","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kamalprit","family":"Chokar","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicholas T.","family":"Vozoris","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marianne S.","family":"Clancy","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marie E.","family":"Faughnan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2025,3,1]]},"reference":[{"issue":"12","key":"3561_CR1","doi-asserted-by":"publisher","first-page":"989","DOI":"10.7326\/m20-1443","volume":"173","author":"ME Faughnan","year":"2020","unstructured":"Faughnan ME, Mager JJ, Hetts SW, Palda VA, Lang-Robertson K, Buscarini E, et al. Second international guidelines for the diagnosis and management of hereditary hemorrhagic telangiectasia. Ann Intern Med. 2020;173(12):989\u20131001. https:\/\/doi.org\/10.7326\/m20-1443.","journal-title":"Ann Intern Med"},{"issue":"11","key":"3561_CR2","doi-asserted-by":"publisher","first-page":"1031","DOI":"10.1160\/th07-01-0064","volume":"98","author":"L Sulaiman","year":"2007","unstructured":"Sulaiman L, Govani F, Jackson J, Begbie M, Shovlin C. Elevated factor VIII in hereditary haemorrhagic telangiectasia (HHT): Association with venous thromboembolism. Thromb Haemost. 2007;98(11):1031\u20139. https:\/\/doi.org\/10.1160\/th07-01-0064.","journal-title":"Thromb Haemost"},{"issue":"3","key":"3561_CR3","doi-asserted-by":"publisher","first-page":"100742","DOI":"10.1016\/j.cpcardiol.2020.100742","volume":"46","author":"JE G\u00f3mez-Mesa","year":"2021","unstructured":"G\u00f3mez-Mesa JE, Galindo-Coral S, Montes MC, Mu\u00f1oz Martin AJ. Thrombosis and coagulopathy in covid-19. Curr Probl Cardiol. 2021;46(3):100742. https:\/\/doi.org\/10.1016\/j.cpcardiol.2020.100742.","journal-title":"Curr Probl Cardiol"},{"key":"3561_CR4","doi-asserted-by":"publisher","unstructured":"Suppressa P, Pagella F, Lenato GM, Gaetani E, Serio I, Masala MS, et al. Characterization of epidemiological distribution and outcome of COVID-19 in patients with hereditary hemorrhagic telangiectasia: a nationwide retrospective multi-centre study during First Wave in Italy. Orphanet J Rare Dis. 2021;16(1). https:\/\/doi.org\/10.1186\/s13023-021-02000-2.","DOI":"10.1186\/s13023-021-02000-2"},{"issue":"9","key":"3561_CR5","doi-asserted-by":"publisher","first-page":"1884","DOI":"10.3390\/jcm10091884","volume":"10","author":"S Marcos","year":"2021","unstructured":"Marcos S, Albi\u00f1ana V, Recio-Poveda L, Tarazona B, Verde-Gonz\u00e1lez MP, Ojeda-Fern\u00e1ndez L, et al. SARS-COV-2 infection in hereditary hemorrhagic telangiectasia patients suggests less clinical impact than in the general population. J Clin Med. 2021;10(9):1884. https:\/\/doi.org\/10.3390\/jcm10091884.","journal-title":"J Clin Med"},{"key":"3561_CR6","doi-asserted-by":"publisher","unstructured":"Mariano RZ, Pereira MC, Reis F. Hereditary hemorrhagic telangiectasia and covid-19. Rev Soc Bras Med Trop. 2020;53. https:\/\/doi.org\/10.1590\/0037-8682-0785-2020.","DOI":"10.1590\/0037-8682-0785-2020"},{"issue":"1","key":"3561_CR7","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1007\/s10456-020-09755-5","volume":"24","author":"A Riera-Mestre","year":"2020","unstructured":"Riera-Mestre A, Iriarte A, Moreno M, del Castillo R, L\u00f3pez-Wolf D. Angiogenesis, hereditary hemorrhagic telangiectasia and covid-19. Angiogenesis. 2020;24(1):13\u20135. https:\/\/doi.org\/10.1007\/s10456-020-09755-5.","journal-title":"Angiogenesis"},{"issue":"7","key":"3561_CR8","doi-asserted-by":"publisher","first-page":"1799","DOI":"10.1111\/jth.14885","volume":"18","author":"E Gaetani","year":"2020","unstructured":"Gaetani E, Passali GC, Riccioni ME, Tortora A, Pola R, Costamagna G, et al. Hereditary Haemorrhagic Telangiectasia: a disease not to be forgotten during the COVID-19 pandemic. J Thromb Haemost. 2020;18(7):1799\u2013801. https:\/\/doi.org\/10.1111\/jth.14885.","journal-title":"J Thromb Haemost"},{"issue":"4","key":"3561_CR9","doi-asserted-by":"publisher","first-page":"175","DOI":"10.1097\/01.gim.0000132689.25644.7c","volume":"6","author":"P Bayrak-Toydemir","year":"2004","unstructured":"Bayrak-Toydemir P, Mao R, Lewin S, McDonald J. Hereditary hemorrhagic telangiectasia: an overview of diagnosis and management in the molecular era for clinicians. Genet Sci. 2004;6(4):175\u201391. https:\/\/doi.org\/10.1097\/01.gim.0000132689.25644.7c.","journal-title":"Genet Sci"},{"key":"3561_CR10","doi-asserted-by":"publisher","DOI":"10.3410\/f.740702617.793588536","author":"A Harries","year":"2021","unstructured":"Harries A, Takarinda KC. Faculty opinions recommendation of risk of thrombocytopenia and thromboembolism after covid-19 vaccination and SARS-COV-2 positive testing: self-controlled case series study. Fac Opinions\u2013 Post-Publication Peer Rev Biomedical Literature. 2021. https:\/\/doi.org\/10.3410\/f.740702617.793588536.","journal-title":"Fac Opinions\u2013 Post-Publication Peer Rev Biomedical Literature"}],"container-title":["Orphanet Journal of Rare Diseases"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13023-025-03561-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13023-025-03561-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13023-025-03561-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T06:13:31Z","timestamp":1740809611000},"score":1,"resource":{"primary":{"URL":"https:\/\/ojrd.biomedcentral.com\/articles\/10.1186\/s13023-025-03561-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3,1]]},"references-count":10,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2025,12]]}},"alternative-id":["3561"],"URL":"https:\/\/doi.org\/10.1186\/s13023-025-03561-2","relation":{"has-preprint":[{"id-type":"doi","id":"10.21203\/rs.3.rs-3728122\/v1","asserted-by":"object"}]},"ISSN":["1750-1172"],"issn-type":[{"value":"1750-1172","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,3,1]]},"assertion":[{"value":"11 December 2023","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 January 2025","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"1 March 2025","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"This study was conducted in accordance with the ethical standards outlined by the Unity Health Toronto Research Ethics Board and received approval prior to study commencement. All participants provided full consent for the anonymous use of their relevant data.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"N\/A.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"94"}}