Higher prevalence of splenic artery aneurysms in hereditary hemorrhagic telangiectasia: Vascular implications and risk factors
Splenic artery aneurysm (SAA) is a rare but potentially fatal condition. Rupture results in 25% mortality up to 75% in pregnant women with 95% fetal mortality. Brief reports suggest an increased risk of developing SAA in patients with HHT. We analyzed enhanced multidetector CT data in 186 HHT patien...
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| Veröffentlicht in: | PloS one 2020-01, Vol.15 (1), p.e0226681-e0226681 |
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| creator | Sellier, Jacques Karam, Carma Beauchet, Alain Dallongeville, Axel Binsse, Stephen Blivet, Sandra Bourgault-Villada, Isabelle Charron, Philippe Chinet, Thierry Eyries, Mélanie Fagnou, Carole Lesniak, Jérome Lesur, Gilles Lucas, Jérome Nicod-Tran, Agnès Ozanne, Augustin Palmyre, Aurélien Soubrier, Florent El Hajjam, Mostafa Lacombe, Pascal |
| description | Splenic artery aneurysm (SAA) is a rare but potentially fatal condition. Rupture results in 25% mortality up to 75% in pregnant women with 95% fetal mortality. Brief reports suggest an increased risk of developing SAA in patients with HHT.
We analyzed enhanced multidetector CT data in 186 HHT patients matched (gender and ± 5 year old) with 186 controls. We screened for SAA and recorded diameter of splenic and hepatic arteries and hepatic, pancreatic and splenic parenchymal involvements. We determined by univariate and multivariate analysis, the relationship with age, sex, genetic status, cardiovascular risk factors (CVRF) and visceral involvement.
SAA concerned 24.7% of HHT patients and 5.4% of controls, p |
| doi_str_mv | 10.1371/journal.pone.0226681 |
| format | Article |
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We analyzed enhanced multidetector CT data in 186 HHT patients matched (gender and ± 5 year old) with 186 controls. We screened for SAA and recorded diameter of splenic and hepatic arteries and hepatic, pancreatic and splenic parenchymal involvements. We determined by univariate and multivariate analysis, the relationship with age, sex, genetic status, cardiovascular risk factors (CVRF) and visceral involvement.
SAA concerned 24.7% of HHT patients and 5.4% of controls, p<0.001. Factors associated with increased risk of SAA in HHT were female gender (p = 0.04, OR = 2.12, IC 95% = 1.03-4.50), age (p = 0.0003, OR = 1.04, 95% CI = 1.02-1.06) and pancreatic parenchymal involvement (p = 0.04, OR = 2.13, 95% CI = 1.01-4.49), but not type of mutation, hepatic or splenic parenchymal involvements, splenic size or splenic artery diameter or CVRF.
We found a 4.57 higher rate of SAA in HHT patients without evidence of splenic high output related disease or increased CVRF. These results suggest the presence of a vascular intrinsic involvement. It should lead to screening all HHT patients for SAA. The vasculopathy hypothesis could require a change in management as screening of all systemic arteries and even the aorta and to further research in the field.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0226681</identifier><identifier>PMID: 31971937</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Adult ; Aged ; Aneurysm ; Aneurysm - diagnostic imaging ; Aneurysm - epidemiology ; Aneurysm - etiology ; Aneurysm - pathology ; Aorta ; Arteries ; Biology and Life Sciences ; Cardiovascular diseases ; Case-Control Studies ; Family medical history ; Female ; Fetuses ; Follow-Up Studies ; France - epidemiology ; Gender ; Genetics ; Health aspects ; Health risks ; Hemorrhage ; Hepatic artery ; Hereditary hemorrhagic telangiectasia ; Homogenization ; Humans ; Hypertension ; Life Sciences ; Liver ; Male ; Medicine and Health Sciences ; Middle Aged ; Mortality ; Multidetector Computed Tomography ; Multivariate analysis ; Mutation ; Pancreas ; Pregnancy ; Pregnant women ; Prevalence ; Prognosis ; Pulmonary arteries ; Research and Analysis Methods ; Retrospective Studies ; Risk analysis ; Risk Factors ; Santé publique et épidémiologie ; Spleen ; Splenic artery ; Splenic Artery - pathology ; Telangiectasia, Hereditary Hemorrhagic - complications ; Telangiectasis ; Vascular diseases ; Vascular Diseases - diagnostic imaging ; Vascular Diseases - epidemiology ; Vascular Diseases - etiology ; Vascular Diseases - pathology ; Veins & arteries ; Women</subject><ispartof>PloS one, 2020-01, Vol.15 (1), p.e0226681-e0226681</ispartof><rights>COPYRIGHT 2020 Public Library of Science</rights><rights>2020 Sellier et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>Distributed under a Creative Commons Attribution 4.0 International License</rights><rights>2020 Sellier et al 2020 Sellier et al</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c726t-45be0cfb728b57f982ddefcd0ff45449436b25aadefa572a1d5c63fa95667a7a3</citedby><cites>FETCH-LOGICAL-c726t-45be0cfb728b57f982ddefcd0ff45449436b25aadefa572a1d5c63fa95667a7a3</cites><orcidid>0000-0001-8863-9201 ; 0000-0002-5470-4367 ; 0000-0003-2571-7932 ; 0000-0003-1911-6698 ; 0000-0002-1782-1118</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC6977744/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC6977744/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,727,780,784,864,885,2102,2928,23866,27924,27925,53791,53793,79600,79601</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/31971937$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink><backlink>$$Uhttps://hal.sorbonne-universite.fr/hal-02945460$$DView record in HAL$$Hfree_for_read</backlink></links><search><contributor>Brusgaard, Klaus</contributor><creatorcontrib>Sellier, Jacques</creatorcontrib><creatorcontrib>Karam, Carma</creatorcontrib><creatorcontrib>Beauchet, Alain</creatorcontrib><creatorcontrib>Dallongeville, Axel</creatorcontrib><creatorcontrib>Binsse, Stephen</creatorcontrib><creatorcontrib>Blivet, Sandra</creatorcontrib><creatorcontrib>Bourgault-Villada, Isabelle</creatorcontrib><creatorcontrib>Charron, Philippe</creatorcontrib><creatorcontrib>Chinet, Thierry</creatorcontrib><creatorcontrib>Eyries, Mélanie</creatorcontrib><creatorcontrib>Fagnou, Carole</creatorcontrib><creatorcontrib>Lesniak, Jérome</creatorcontrib><creatorcontrib>Lesur, Gilles</creatorcontrib><creatorcontrib>Lucas, Jérome</creatorcontrib><creatorcontrib>Nicod-Tran, Agnès</creatorcontrib><creatorcontrib>Ozanne, Augustin</creatorcontrib><creatorcontrib>Palmyre, Aurélien</creatorcontrib><creatorcontrib>Soubrier, Florent</creatorcontrib><creatorcontrib>El Hajjam, Mostafa</creatorcontrib><creatorcontrib>Lacombe, Pascal</creatorcontrib><title>Higher prevalence of splenic artery aneurysms in hereditary hemorrhagic telangiectasia: Vascular implications and risk factors</title><title>PloS one</title><addtitle>PLoS One</addtitle><description>Splenic artery aneurysm (SAA) is a rare but potentially fatal condition. Rupture results in 25% mortality up to 75% in pregnant women with 95% fetal mortality. Brief reports suggest an increased risk of developing SAA in patients with HHT.
We analyzed enhanced multidetector CT data in 186 HHT patients matched (gender and ± 5 year old) with 186 controls. We screened for SAA and recorded diameter of splenic and hepatic arteries and hepatic, pancreatic and splenic parenchymal involvements. We determined by univariate and multivariate analysis, the relationship with age, sex, genetic status, cardiovascular risk factors (CVRF) and visceral involvement.
SAA concerned 24.7% of HHT patients and 5.4% of controls, p<0.001. Factors associated with increased risk of SAA in HHT were female gender (p = 0.04, OR = 2.12, IC 95% = 1.03-4.50), age (p = 0.0003, OR = 1.04, 95% CI = 1.02-1.06) and pancreatic parenchymal involvement (p = 0.04, OR = 2.13, 95% CI = 1.01-4.49), but not type of mutation, hepatic or splenic parenchymal involvements, splenic size or splenic artery diameter or CVRF.
We found a 4.57 higher rate of SAA in HHT patients without evidence of splenic high output related disease or increased CVRF. These results suggest the presence of a vascular intrinsic involvement. It should lead to screening all HHT patients for SAA. The vasculopathy hypothesis could require a change in management as screening of all systemic arteries and even the aorta and to further research in the field.</description><subject>Adult</subject><subject>Aged</subject><subject>Aneurysm</subject><subject>Aneurysm - diagnostic imaging</subject><subject>Aneurysm - epidemiology</subject><subject>Aneurysm - etiology</subject><subject>Aneurysm - pathology</subject><subject>Aorta</subject><subject>Arteries</subject><subject>Biology and Life Sciences</subject><subject>Cardiovascular diseases</subject><subject>Case-Control Studies</subject><subject>Family medical history</subject><subject>Female</subject><subject>Fetuses</subject><subject>Follow-Up Studies</subject><subject>France - epidemiology</subject><subject>Gender</subject><subject>Genetics</subject><subject>Health aspects</subject><subject>Health risks</subject><subject>Hemorrhage</subject><subject>Hepatic artery</subject><subject>Hereditary hemorrhagic telangiectasia</subject><subject>Homogenization</subject><subject>Humans</subject><subject>Hypertension</subject><subject>Life Sciences</subject><subject>Liver</subject><subject>Male</subject><subject>Medicine and Health Sciences</subject><subject>Middle Aged</subject><subject>Mortality</subject><subject>Multidetector Computed Tomography</subject><subject>Multivariate analysis</subject><subject>Mutation</subject><subject>Pancreas</subject><subject>Pregnancy</subject><subject>Pregnant women</subject><subject>Prevalence</subject><subject>Prognosis</subject><subject>Pulmonary arteries</subject><subject>Research and Analysis Methods</subject><subject>Retrospective Studies</subject><subject>Risk analysis</subject><subject>Risk Factors</subject><subject>Santé publique et épidémiologie</subject><subject>Spleen</subject><subject>Splenic artery</subject><subject>Splenic Artery - pathology</subject><subject>Telangiectasia, Hereditary Hemorrhagic - complications</subject><subject>Telangiectasis</subject><subject>Vascular diseases</subject><subject>Vascular Diseases - diagnostic imaging</subject><subject>Vascular Diseases - epidemiology</subject><subject>Vascular Diseases - etiology</subject><subject>Vascular Diseases - pathology</subject><subject>Veins & arteries</subject><subject>Women</subject><issn>1932-6203</issn><issn>1932-6203</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><sourceid>DOA</sourceid><recordid>eNqNk01v1DAQhiMEoqXwDxBEQkL0sIvtOHbCAWlVAbvSSpX46NWa2E7iJYm3drKiF3473m5abaoeUA6xXj_z2jOeiaLXGM1xwvHHjR1cB818azs9R4QwluEn0SnOEzJjBCVPj9Yn0QvvNwilScbY8-gkwTkPm_w0-rs0Va1dvHV6B43upI5tGfttWBoZg-u1u4mh04O78a2PTRcHWivTQ9Br3VrnaqgC2usGuspo2YM38Cm-Ai-HBlxs2m1jJPTGdj44qdgZ_zsuQfbW-ZfRsxIar1-N_7Po19cvPy-Ws_Xlt9XFYj2TnLB-RtNCI1kWnGRFyss8I0rpUipUljSlNKcJK0gKEERIOQGsUsmSEvKUMQ4ckrPo7cF321gvxtJ5QRJKCeIZ4YFYHQhlYSO2zrQhQ2HBiFvBukqEahjZaJEWmCtFKVcZpgmgnJAESyTzTIEiOQlen8fThqLVSuqud9BMTKc7nalFZXeC5ZxzSoPB-cGgfhC2XKzFXkMkD4kztMOB_TAe5uz1oH0vWuOlbsJzaDuMOXKSkj367gH6eCVGqgoNIUxX2nBHuTcVC4YJyrIc7XOcP0KFT-nWyNCTpQn6JOB8EhCYXv_pKxi8F6sf3_-fvbyasu-P2FpD09feNsNtw01BegCls947Xd5XFiOxH6m7aoj9SIlxpELYm-PHvA-6m6HkH4-zHYQ</recordid><startdate>20200123</startdate><enddate>20200123</enddate><creator>Sellier, 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prevalence of splenic artery aneurysms in hereditary hemorrhagic telangiectasia: Vascular implications and risk factors</title><author>Sellier, Jacques ; Karam, Carma ; Beauchet, Alain ; Dallongeville, Axel ; Binsse, Stephen ; Blivet, Sandra ; Bourgault-Villada, Isabelle ; Charron, Philippe ; Chinet, Thierry ; Eyries, Mélanie ; Fagnou, Carole ; Lesniak, Jérome ; Lesur, Gilles ; Lucas, Jérome ; Nicod-Tran, Agnès ; Ozanne, Augustin ; Palmyre, Aurélien ; Soubrier, Florent ; El Hajjam, Mostafa ; Lacombe, Pascal</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c726t-45be0cfb728b57f982ddefcd0ff45449436b25aadefa572a1d5c63fa95667a7a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Adult</topic><topic>Aged</topic><topic>Aneurysm</topic><topic>Aneurysm - diagnostic imaging</topic><topic>Aneurysm - epidemiology</topic><topic>Aneurysm - etiology</topic><topic>Aneurysm - 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Academic</collection><collection>ProQuest Engineering Collection</collection><collection>ProQuest Biological Science Collection</collection><collection>Agricultural Science Database</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Algology Mycology and Protozoology Abstracts (Microbiology C)</collection><collection>Biological Science Database</collection><collection>Engineering Database</collection><collection>Nursing & Allied Health Premium</collection><collection>Advanced Technologies & Aerospace Database</collection><collection>ProQuest Advanced Technologies & Aerospace Collection</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Environmental Science Database</collection><collection>Materials Science Collection</collection><collection>Access via ProQuest (Open Access)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>Engineering Collection</collection><collection>Environmental Science Collection</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>Hyper Article en Ligne (HAL)</collection><collection>Hyper Article en Ligne (HAL) (Open Access)</collection><collection>PubMed Central (Full Participant titles)</collection><collection>DOAJ Directory of Open Access Journals</collection><jtitle>PloS one</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Sellier, Jacques</au><au>Karam, Carma</au><au>Beauchet, Alain</au><au>Dallongeville, Axel</au><au>Binsse, Stephen</au><au>Blivet, Sandra</au><au>Bourgault-Villada, Isabelle</au><au>Charron, Philippe</au><au>Chinet, Thierry</au><au>Eyries, Mélanie</au><au>Fagnou, Carole</au><au>Lesniak, Jérome</au><au>Lesur, Gilles</au><au>Lucas, Jérome</au><au>Nicod-Tran, Agnès</au><au>Ozanne, Augustin</au><au>Palmyre, Aurélien</au><au>Soubrier, Florent</au><au>El Hajjam, Mostafa</au><au>Lacombe, Pascal</au><au>Brusgaard, Klaus</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Higher prevalence of splenic artery aneurysms in hereditary hemorrhagic telangiectasia: Vascular implications and risk factors</atitle><jtitle>PloS one</jtitle><addtitle>PLoS One</addtitle><date>2020-01-23</date><risdate>2020</risdate><volume>15</volume><issue>1</issue><spage>e0226681</spage><epage>e0226681</epage><pages>e0226681-e0226681</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>Splenic artery aneurysm (SAA) is a rare but potentially fatal condition. Rupture results in 25% mortality up to 75% in pregnant women with 95% fetal mortality. Brief reports suggest an increased risk of developing SAA in patients with HHT.
We analyzed enhanced multidetector CT data in 186 HHT patients matched (gender and ± 5 year old) with 186 controls. We screened for SAA and recorded diameter of splenic and hepatic arteries and hepatic, pancreatic and splenic parenchymal involvements. We determined by univariate and multivariate analysis, the relationship with age, sex, genetic status, cardiovascular risk factors (CVRF) and visceral involvement.
SAA concerned 24.7% of HHT patients and 5.4% of controls, p<0.001. Factors associated with increased risk of SAA in HHT were female gender (p = 0.04, OR = 2.12, IC 95% = 1.03-4.50), age (p = 0.0003, OR = 1.04, 95% CI = 1.02-1.06) and pancreatic parenchymal involvement (p = 0.04, OR = 2.13, 95% CI = 1.01-4.49), but not type of mutation, hepatic or splenic parenchymal involvements, splenic size or splenic artery diameter or CVRF.
We found a 4.57 higher rate of SAA in HHT patients without evidence of splenic high output related disease or increased CVRF. These results suggest the presence of a vascular intrinsic involvement. It should lead to screening all HHT patients for SAA. The vasculopathy hypothesis could require a change in management as screening of all systemic arteries and even the aorta and to further research in the field.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>31971937</pmid><doi>10.1371/journal.pone.0226681</doi><tpages>e0226681</tpages><orcidid>https://orcid.org/0000-0001-8863-9201</orcidid><orcidid>https://orcid.org/0000-0002-5470-4367</orcidid><orcidid>https://orcid.org/0000-0003-2571-7932</orcidid><orcidid>https://orcid.org/0000-0003-1911-6698</orcidid><orcidid>https://orcid.org/0000-0002-1782-1118</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 1932-6203 |
| ispartof | PloS one, 2020-01, Vol.15 (1), p.e0226681-e0226681 |
| issn | 1932-6203 1932-6203 |
| language | eng |
| recordid | cdi_plos_journals_2344207827 |
| source | MEDLINE; DOAJ Directory of Open Access Journals; Public Library of Science (PLoS) Journals Open Access; EZB-FREE-00999 freely available EZB journals; PubMed Central; Free Full-Text Journals in Chemistry |
| subjects | Adult Aged Aneurysm Aneurysm - diagnostic imaging Aneurysm - epidemiology Aneurysm - etiology Aneurysm - pathology Aorta Arteries Biology and Life Sciences Cardiovascular diseases Case-Control Studies Family medical history Female Fetuses Follow-Up Studies France - epidemiology Gender Genetics Health aspects Health risks Hemorrhage Hepatic artery Hereditary hemorrhagic telangiectasia Homogenization Humans Hypertension Life Sciences Liver Male Medicine and Health Sciences Middle Aged Mortality Multidetector Computed Tomography Multivariate analysis Mutation Pancreas Pregnancy Pregnant women Prevalence Prognosis Pulmonary arteries Research and Analysis Methods Retrospective Studies Risk analysis Risk Factors Santé publique et épidémiologie Spleen Splenic artery Splenic Artery - pathology Telangiectasia, Hereditary Hemorrhagic - complications Telangiectasis Vascular diseases Vascular Diseases - diagnostic imaging Vascular Diseases - epidemiology Vascular Diseases - etiology Vascular Diseases - pathology Veins & arteries Women |
| title | Higher prevalence of splenic artery aneurysms in hereditary hemorrhagic telangiectasia: Vascular implications and risk factors |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-28T13%3A59%3A20IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-gale_plos_&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Higher%20prevalence%20of%20splenic%20artery%20aneurysms%20in%20hereditary%20hemorrhagic%20telangiectasia:%20Vascular%20implications%20and%20risk%20factors&rft.jtitle=PloS%20one&rft.au=Sellier,%20Jacques&rft.date=2020-01-23&rft.volume=15&rft.issue=1&rft.spage=e0226681&rft.epage=e0226681&rft.pages=e0226681-e0226681&rft.issn=1932-6203&rft.eissn=1932-6203&rft_id=info:doi/10.1371/journal.pone.0226681&rft_dat=%3Cgale_plos_%3EA612088902%3C/gale_plos_%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2344207827&rft_id=info:pmid/31971937&rft_galeid=A612088902&rft_doaj_id=oai_doaj_org_article_5b17dd447d8143a092231c0c98dad292&rfr_iscdi=true |