Cardiovascular Events in Patients With Fabry Disease: Natural History Data From the Fabry Registry
Identifieur interne : 006C81 ( Main/Exploration ); précédent : 006C80; suivant : 006C82Cardiovascular Events in Patients With Fabry Disease: Natural History Data From the Fabry Registry
Auteurs : Manesh R. Patel [États-Unis] ; Franco Cecchi [Italie] ; Marta Cizmarik [États-Unis] ; Ilkka Kantola [Finlande] ; Ales Linhart [République tchèque] ; Kathy Nicholls [Australie] ; Jörg Strotmann [Allemagne] ; Jose Tallaj [États-Unis] ; THI CHIEN TRAN [France] ; Michael L. West [Canada] ; Dana Beitner-Johnson [Italie] ; Ademola Abiose [États-Unis]Source :
- Journal of the American College of Cardiology [ 0735-1097 ] ; 2011.
Descripteurs français
- Pascal (Inist)
- Wicri :
- topic : Homme, Histoire, Historique.
English descriptors
- KwdEn :
Abstract
Objectives These analyses were designed to determine the incidence of major cardiovascular (CV) events and the natural history of CV complications in patients with Fabry disease. Background Fabry disease, a genetic disorder caused by deficiency of alpha-galactosidase A activity, is associated with CV dysfunction. Methods Major CV events (myocardial infarction, heart failure, or cardiac-related death) were analyzed in 2,869 Fabry Registry patients during the natural history period (i.e., before enzyme replacement therapy or among patients who never received therapy). Multivariate logistic regression analyses were performed to identify significant predictors of CV events. Results Eighty-three of 1,424 men (5.8%) and 54 of 1,445 women (3.7%) experienced CV events at mean ages of 45 and 54 years, respectively. Heart failure was the most common first CV event, reported by 50 men (3.5%) and 33 women (2.3%). Hypertension and left ventricular hypertrophy were the risk factors most strongly associated with CV events. When these parameters were used as covariates in logistic regression analyses, the odds ratio (OR) for hypertension in men was 7.8 (95% confidence interval [CI]: 2.1 to 28.6, p = 0.0019), and the OR for hypertension in women was 4.5 (95% CI: 1.6 to 12.3, p = 0.0037). The OR for left ventricular hypertrophy was 4.8 in men (95% CI: 1.03 to 22.2, p = 0.0463) and 8.2 in women (95% CI: 2.6 to 26.0, p = 0.0003). Conclusions Major CV events occurred in approximately 5% of Fabry Registry patients during the natural history period. All patients with Fabry disease should be monitored for possible CV risk factors, particularly hypertension and left ventricular hypertrophy.
Affiliations:
- Allemagne, Australie, Canada, Finlande, France, Italie, République tchèque, États-Unis
- Alabama, Bohême centrale, Caroline du Nord, Finlande occidentale, Iowa, Massachusetts, Schleswig-Holstein, Victoria (État)
- Iowa City, Kiel, Melbourne, Prague, Turku
- Université de Melbourne, Université de l'Iowa
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West</name><affiliation wicri:level="1"><inist:fA14 i1="10"><s1>Division of Nephrology, Department of Medicine, Dalhousie University</s1><s2>Halifax, Nova Scotia</s2><s3>CAN</s3><sZ>10 aut.</sZ></inist:fA14><country>Canada</country><wicri:noRegion>Halifax, Nova Scotia</wicri:noRegion></affiliation></author><author><name sortKey="Beitner Johnson, Dana" sort="Beitner Johnson, Dana" uniqKey="Beitner Johnson D" first="Dana" last="Beitner-Johnson">Dana Beitner-Johnson</name><affiliation wicri:level="1"><inist:fA14 i1="02"><s1>Cardiology Department, Careggi University Hospital</s1><s2>Firenze</s2><s3>ITA</s3><sZ>2 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Firenze</wicri:noRegion></affiliation></author><author><name sortKey="Abiose, Ademola" sort="Abiose, Ademola" uniqKey="Abiose A" first="Ademola" last="Abiose">Ademola Abiose</name><affiliation wicri:level="4"><inist:fA14 i1="11"><s1>Division of Cardiovascular Medicine, University of Iowa</s1><s2>Iowa City, Iowa</s2><s3>USA</s3><sZ>12 aut.</sZ></inist:fA14><country>États-Unis</country><placeName><region type="state">Iowa</region><settlement type="city">Iowa City</settlement></placeName><orgName type="university">Université de l'Iowa</orgName></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">INIST</idno><idno type="inist">11-0150626</idno><date when="2011">2011</date><idno type="stanalyst">PASCAL 11-0150626 INIST</idno><idno type="RBID">Pascal:11-0150626</idno><idno type="wicri:Area/PascalFrancis/Corpus">001F28</idno><idno type="wicri:Area/PascalFrancis/Curation">004008</idno><idno type="wicri:Area/PascalFrancis/Checkpoint">001C89</idno><idno type="wicri:explorRef" wicri:stream="PascalFrancis" wicri:step="Checkpoint">001C89</idno><idno type="wicri:doubleKey">0735-1097:2011:Patel M:cardiovascular:events:in</idno><idno type="wicri:Area/Main/Merge">007140</idno><idno type="wicri:Area/Main/Curation">006C81</idno><idno type="wicri:Area/Main/Exploration">006C81</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a">Cardiovascular Events in Patients With Fabry Disease: Natural History Data From the Fabry Registry</title><author><name sortKey="Patel, Manesh R" sort="Patel, Manesh R" uniqKey="Patel M" first="Manesh R." last="Patel">Manesh R. Patel</name><affiliation wicri:level="2"><inist:fA14 i1="01"><s1>Division of Cardiovascular Medicine, Duke University School of Medicine</s1><s2>Durham, North Carolina</s2><s3>USA</s3><sZ>1 aut.</sZ></inist:fA14><country>États-Unis</country><placeName><region type="state">Caroline du Nord</region></placeName></affiliation></author><author><name sortKey="Cecchi, Franco" sort="Cecchi, Franco" uniqKey="Cecchi F" first="Franco" last="Cecchi">Franco Cecchi</name><affiliation wicri:level="1"><inist:fA14 i1="02"><s1>Cardiology Department, Careggi University Hospital</s1><s2>Firenze</s2><s3>ITA</s3><sZ>2 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Firenze</wicri:noRegion></affiliation></author><author><name sortKey="Cizmarik, Marta" sort="Cizmarik, Marta" uniqKey="Cizmarik M" first="Marta" last="Cizmarik">Marta Cizmarik</name><affiliation wicri:level="2"><inist:fA14 i1="03"><s1>Biomedical Data Sciences and Informatics, Genzyme Corporation</s1><s2>Cambridge, Massachusetts</s2><s3>USA</s3><sZ>3 aut.</sZ></inist:fA14><country>États-Unis</country><placeName><region type="state">Massachusetts</region></placeName></affiliation></author><author><name sortKey="Kantola, Ilkka" sort="Kantola, Ilkka" uniqKey="Kantola I" first="Ilkka" last="Kantola">Ilkka Kantola</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Turku University Hospital, Department of Medicine, University of Turku</s1><s2>Turku</s2><s3>FIN</s3><sZ>4 aut.</sZ></inist:fA14><country>Finlande</country><placeName><settlement type="city">Turku</settlement><region type="région" nuts="2">Finlande occidentale</region></placeName></affiliation></author><author><name sortKey="Linhart, Ales" sort="Linhart, Ales" uniqKey="Linhart A" first="Ales" last="Linhart">Ales Linhart</name><affiliation wicri:level="3"><inist:fA14 i1="05"><s1>Department of Internal Medicine, School of Medicine, Charles University</s1><s2>Prague</s2><s3>CZE</s3><sZ>5 aut.</sZ></inist:fA14><country>République tchèque</country><placeName><settlement type="city">Prague</settlement><region type="région" nuts="2">Bohême centrale</region></placeName></affiliation></author><author><name sortKey="Nicholls, Kathy" sort="Nicholls, Kathy" uniqKey="Nicholls K" first="Kathy" last="Nicholls">Kathy Nicholls</name><affiliation wicri:level="4"><inist:fA14 i1="06"><s1>Department of Nephrology, Royal Melbourne Hospital and University of Melbourne</s1><s2>Parkville</s2><s3>AUS</s3><sZ>6 aut.</sZ></inist:fA14><country>Australie</country><placeName><settlement type="city">Melbourne</settlement><region type="état">Victoria (État)</region></placeName><orgName type="university">Université de Melbourne</orgName></affiliation></author><author><name sortKey="Strotmann, Jorg" sort="Strotmann, Jorg" uniqKey="Strotmann J" first="Jörg" last="Strotmann">Jörg Strotmann</name><affiliation wicri:level="3"><inist:fA14 i1="07"><s1>Medizinische Klinik, Stadtisches Krankenhaus</s1><s2>Kiel</s2><s3>DEU</s3><sZ>7 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="2">Schleswig-Holstein</region><settlement type="city">Kiel</settlement></placeName></affiliation></author><author><name sortKey="Tallaj, Jose" sort="Tallaj, Jose" uniqKey="Tallaj J" first="Jose" last="Tallaj">Jose Tallaj</name><affiliation wicri:level="2"><inist:fA14 i1="08"><s1>Department of Cardiovascular Disease, University of Alabama at Birmingham</s1><s2>Birmingham, Alabama</s2><s3>USA</s3><sZ>8 aut.</sZ></inist:fA14><country>États-Unis</country><placeName><region type="state">Alabama</region></placeName></affiliation></author><author><name sortKey="Thi Chien Tran" sort="Thi Chien Tran" uniqKey="Thi Chien Tran" last="Thi Chien Tran">THI CHIEN TRAN</name><affiliation wicri:level="1"><inist:fA14 i1="09"><s1>Division of Medical Genetics, Hôpital Raymond Poincare (AP-HP), University of Versailles-St. Quentin en Yvelines</s1><s2>Garches</s2><s3>FRA</s3><sZ>9 aut.</sZ></inist:fA14><country>France</country><wicri:noRegion>Garches</wicri:noRegion><wicri:noRegion>University of Versailles-St. Quentin en Yvelines</wicri:noRegion><wicri:noRegion>Garches</wicri:noRegion></affiliation></author><author><name sortKey="West, Michael L" sort="West, Michael L" uniqKey="West M" first="Michael L." last="West">Michael L. West</name><affiliation wicri:level="1"><inist:fA14 i1="10"><s1>Division of Nephrology, Department of Medicine, Dalhousie University</s1><s2>Halifax, Nova Scotia</s2><s3>CAN</s3><sZ>10 aut.</sZ></inist:fA14><country>Canada</country><wicri:noRegion>Halifax, Nova Scotia</wicri:noRegion></affiliation></author><author><name sortKey="Beitner Johnson, Dana" sort="Beitner Johnson, Dana" uniqKey="Beitner Johnson D" first="Dana" last="Beitner-Johnson">Dana Beitner-Johnson</name><affiliation wicri:level="1"><inist:fA14 i1="02"><s1>Cardiology Department, Careggi University Hospital</s1><s2>Firenze</s2><s3>ITA</s3><sZ>2 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Firenze</wicri:noRegion></affiliation></author><author><name sortKey="Abiose, Ademola" sort="Abiose, Ademola" uniqKey="Abiose A" first="Ademola" last="Abiose">Ademola Abiose</name><affiliation wicri:level="4"><inist:fA14 i1="11"><s1>Division of Cardiovascular Medicine, University of Iowa</s1><s2>Iowa City, Iowa</s2><s3>USA</s3><sZ>12 aut.</sZ></inist:fA14><country>États-Unis</country><placeName><region type="state">Iowa</region><settlement type="city">Iowa City</settlement></placeName><orgName type="university">Université de l'Iowa</orgName></affiliation></author></analytic><series><title level="j" type="main">Journal of the American College of Cardiology</title><title level="j" type="abbreviated">J. Am. Coll. Cardiol.</title><idno type="ISSN">0735-1097</idno><imprint><date when="2011">2011</date></imprint></series></biblStruct></sourceDesc><seriesStmt><title level="j" type="main">Journal of the American College of Cardiology</title><title level="j" type="abbreviated">J. Am. Coll. Cardiol.</title><idno type="ISSN">0735-1097</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Cardiology</term><term>Case history</term><term>Circulatory system</term><term>Data</term><term>Fabry disease</term><term>History</term><term>Human</term><term>Lipids</term><term>Natural</term><term>Patient</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Sphingolipidose héréditaire de Fabry</term><term>Homme</term><term>Malade</term><term>Naturel</term><term>Histoire</term><term>Historique</term><term>Donnée</term><term>Appareil circulatoire</term><term>Cardiologie</term><term>Lipide</term></keywords><keywords scheme="Wicri" type="topic" xml:lang="fr"><term>Homme</term><term>Histoire</term><term>Historique</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Objectives These analyses were designed to determine the incidence of major cardiovascular (CV) events and the natural history of CV complications in patients with Fabry disease. Background Fabry disease, a genetic disorder caused by deficiency of alpha-galactosidase A activity, is associated with CV dysfunction. Methods Major CV events (myocardial infarction, heart failure, or cardiac-related death) were analyzed in 2,869 Fabry Registry patients during the natural history period (i.e., before enzyme replacement therapy or among patients who never received therapy). Multivariate logistic regression analyses were performed to identify significant predictors of CV events. Results Eighty-three of 1,424 men (5.8%) and 54 of 1,445 women (3.7%) experienced CV events at mean ages of 45 and 54 years, respectively. Heart failure was the most common first CV event, reported by 50 men (3.5%) and 33 women (2.3%). Hypertension and left ventricular hypertrophy were the risk factors most strongly associated with CV events. When these parameters were used as covariates in logistic regression analyses, the odds ratio (OR) for hypertension in men was 7.8 (95% confidence interval [CI]: 2.1 to 28.6, p = 0.0019), and the OR for hypertension in women was 4.5 (95% CI: 1.6 to 12.3, p = 0.0037). The OR for left ventricular hypertrophy was 4.8 in men (95% CI: 1.03 to 22.2, p = 0.0463) and 8.2 in women (95% CI: 2.6 to 26.0, p = 0.0003). Conclusions Major CV events occurred in approximately 5% of Fabry Registry patients during the natural history period. All patients with Fabry disease should be monitored for possible CV risk factors, particularly hypertension and left ventricular hypertrophy.</div></front></TEI><affiliations><list><country><li>Allemagne</li><li>Australie</li><li>Canada</li><li>Finlande</li><li>France</li><li>Italie</li><li>République tchèque</li><li>États-Unis</li></country><region><li>Alabama</li><li>Bohême centrale</li><li>Caroline du Nord</li><li>Finlande occidentale</li><li>Iowa</li><li>Massachusetts</li><li>Schleswig-Holstein</li><li>Victoria (État)</li></region><settlement><li>Iowa City</li><li>Kiel</li><li>Melbourne</li><li>Prague</li><li>Turku</li></settlement><orgName><li>Université de Melbourne</li><li>Université de l'Iowa</li></orgName></list><tree><country name="États-Unis"><region name="Caroline du Nord"><name sortKey="Patel, Manesh R" sort="Patel, Manesh R" uniqKey="Patel M" first="Manesh R." last="Patel">Manesh R. Patel</name></region><name sortKey="Abiose, Ademola" sort="Abiose, Ademola" uniqKey="Abiose A" first="Ademola" last="Abiose">Ademola Abiose</name><name sortKey="Cizmarik, Marta" sort="Cizmarik, Marta" uniqKey="Cizmarik M" first="Marta" last="Cizmarik">Marta Cizmarik</name><name sortKey="Tallaj, Jose" sort="Tallaj, Jose" uniqKey="Tallaj J" first="Jose" last="Tallaj">Jose Tallaj</name></country><country name="Italie"><noRegion><name sortKey="Cecchi, Franco" sort="Cecchi, Franco" uniqKey="Cecchi F" first="Franco" last="Cecchi">Franco Cecchi</name></noRegion><name sortKey="Beitner Johnson, Dana" sort="Beitner Johnson, Dana" uniqKey="Beitner Johnson D" first="Dana" last="Beitner-Johnson">Dana Beitner-Johnson</name></country><country name="Finlande"><region name="Finlande occidentale"><name sortKey="Kantola, Ilkka" sort="Kantola, Ilkka" uniqKey="Kantola I" first="Ilkka" last="Kantola">Ilkka Kantola</name></region></country><country name="République tchèque"><region name="Bohême centrale"><name sortKey="Linhart, Ales" sort="Linhart, Ales" uniqKey="Linhart A" first="Ales" last="Linhart">Ales Linhart</name></region></country><country name="Australie"><region name="Victoria (État)"><name sortKey="Nicholls, Kathy" sort="Nicholls, Kathy" uniqKey="Nicholls K" first="Kathy" last="Nicholls">Kathy Nicholls</name></region></country><country name="Allemagne"><region name="Schleswig-Holstein"><name sortKey="Strotmann, Jorg" sort="Strotmann, Jorg" uniqKey="Strotmann J" first="Jörg" last="Strotmann">Jörg Strotmann</name></region></country><country name="France"><noRegion><name sortKey="Thi Chien Tran" sort="Thi Chien Tran" uniqKey="Thi Chien Tran" last="Thi Chien Tran">THI CHIEN TRAN</name></noRegion></country><country name="Canada"><noRegion><name sortKey="West, Michael L" sort="West, Michael L" uniqKey="West M" first="Michael L." last="West">Michael L. 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