Hyper-IgE syndrome and autoimmunity in Mexican children
Identifieur interne : 002644 ( Istex/Curation ); précédent : 002643; suivant : 002645Hyper-IgE syndrome and autoimmunity in Mexican children
Auteurs : Marco Yamazaki-Nakashimada [Mexique] ; Samuel Zaltzman-Girshevich [Mexique] ; Silvestre Garcia De La Puente [Mexique] ; Beatriz De Leon-Bojorge [Mexique] ; Sara Espinosa-Padilla [Mexique] ; Marimar Saez-De-Ocariz [Mexique] ; Daniel Carrasco-Daza [Mexique] ; Victor Hernandez-Bautista [Mexique] ; Lorenzo Pérez-Fernandez [Mexique] ; Francisco Espinosa-Rosales [Mexique]Source :
- Pediatric Nephrology [ 0931-041X ] ; 2006-08-01.
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Abstract
Abstract: Hyper-IgE syndrome (HIES) is a primary immunodeficiency characterized by recurrent skin abscesses, recurrent pneumonia with pneumatocele formation, eczema, eosinophilia, and elevated levels of serum IgE. Patients with the autosomal recessive (AR) form of HIES appear to be prone to developing autoimmune diseases. We present two cases of HIES with autoimmune complications; one case was a product of a consanguineous marriage, the other one was a sporadic case. The first patient presented with recurrent episodes of erythema nodosum, warts, bronchiolitis obliterans and thrombocytopenia. The second patient developed glomerulonephritis resulting in endstage renal failure. She later developed malar rash, oral ulcers, cerebral infarcts with vasculitis and positive ANA, anti-dsDNA, and antiphospholipid antibodies. We discuss the dilemma in treating patients who present with both primary immunodeficiency and autoimmunity.
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DOI: 10.1007/s00467-006-0178-3
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Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Saez De Ocariz, Marimar" sort="Saez De Ocariz, Marimar" uniqKey="Saez De Ocariz M" first="Marimar" last="Saez-De-Ocariz">Marimar Saez-De-Ocariz</name><affiliation wicri:level="1"><mods:affiliation>Department of Dermatology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Dermatology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. 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Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Perez Fernandez, Lorenzo" sort="Perez Fernandez, Lorenzo" uniqKey="Perez Fernandez L" first="Lorenzo" last="Pérez-Fernandez">Lorenzo Pérez-Fernandez</name><affiliation wicri:level="1"><mods:affiliation>Department of Pulmonology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Pulmonology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. 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Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation><affiliation wicri:level="1"><mods:affiliation>E-mail: yzki71@yahoo.com.mx</mods:affiliation><country wicri:rule="url">Mexique</country></affiliation></author><author><name sortKey="Zaltzman Girshevich, Samuel" sort="Zaltzman Girshevich, Samuel" uniqKey="Zaltzman Girshevich S" first="Samuel" last="Zaltzman-Girshevich">Samuel Zaltzman-Girshevich</name><affiliation wicri:level="1"><mods:affiliation>Department of Nephrology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Nephrology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Garcia De La Puente, Silvestre" sort="Garcia De La Puente, Silvestre" uniqKey="Garcia De La Puente S" first="Silvestre" last="Garcia De La Puente">Silvestre Garcia De La Puente</name><affiliation wicri:level="1"><mods:affiliation>Department of Nephrology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Nephrology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="De Leon Bojorge, Beatriz" sort="De Leon Bojorge, Beatriz" uniqKey="De Leon Bojorge B" first="Beatriz" last="De Leon-Bojorge">Beatriz De Leon-Bojorge</name><affiliation wicri:level="1"><mods:affiliation>Department of Pathology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Pathology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Espinosa Padilla, Sara" sort="Espinosa Padilla, Sara" uniqKey="Espinosa Padilla S" first="Sara" last="Espinosa-Padilla">Sara Espinosa-Padilla</name><affiliation wicri:level="1"><mods:affiliation>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Saez De Ocariz, Marimar" sort="Saez De Ocariz, Marimar" uniqKey="Saez De Ocariz M" first="Marimar" last="Saez-De-Ocariz">Marimar Saez-De-Ocariz</name><affiliation wicri:level="1"><mods:affiliation>Department of Dermatology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Dermatology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Carrasco Daza, Daniel" sort="Carrasco Daza, Daniel" uniqKey="Carrasco Daza D" first="Daniel" last="Carrasco-Daza">Daniel Carrasco-Daza</name><affiliation wicri:level="1"><mods:affiliation>Department of Pathology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Pathology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Hernandez Bautista, Victor" sort="Hernandez Bautista, Victor" uniqKey="Hernandez Bautista V" first="Victor" last="Hernandez-Bautista">Victor Hernandez-Bautista</name><affiliation wicri:level="1"><mods:affiliation>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Perez Fernandez, Lorenzo" sort="Perez Fernandez, Lorenzo" uniqKey="Perez Fernandez L" first="Lorenzo" last="Pérez-Fernandez">Lorenzo Pérez-Fernandez</name><affiliation wicri:level="1"><mods:affiliation>Department of Pulmonology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Pulmonology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author><author><name sortKey="Espinosa Rosales, Francisco" sort="Espinosa Rosales, Francisco" uniqKey="Espinosa Rosales F" first="Francisco" last="Espinosa-Rosales">Francisco Espinosa-Rosales</name><affiliation wicri:level="1"><mods:affiliation>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City, Mexico</mods:affiliation><country xml:lang="fr">Mexique</country><wicri:regionArea>Department of Clinical Immunology, Instituto Nacional de Pediatría, Insurgentes Sur 3700-C, Col. Insurgentes Cuicuilco, Mexico City</wicri:regionArea></affiliation></author></analytic><monogr></monogr><series><title level="j">Pediatric Nephrology</title><title level="j" type="sub">Journal of the International Pediatric Nephrology Association</title><title level="j" type="abbrev">Pediatr Nephrol</title><idno type="ISSN">0931-041X</idno><idno type="eISSN">1432-198X</idno><imprint><publisher>Springer-Verlag</publisher><pubPlace>Berlin/Heidelberg</pubPlace><date type="published" when="2006-08-01">2006-08-01</date><biblScope unit="volume">21</biblScope><biblScope unit="issue">8</biblScope><biblScope unit="page" from="1200">1200</biblScope><biblScope unit="page" to="1205">1205</biblScope></imprint><idno type="ISSN">0931-041X</idno></series></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0931-041X</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Antiphospholipid syndrome</term><term>Autoimmunity</term><term>CNS vasculitis</term><term>Hyper-IgE syndrome</term><term>Primary immunodeficiency</term><term>Systemic lupus erythematosus</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Abstract: Hyper-IgE syndrome (HIES) is a primary immunodeficiency characterized by recurrent skin abscesses, recurrent pneumonia with pneumatocele formation, eczema, eosinophilia, and elevated levels of serum IgE. Patients with the autosomal recessive (AR) form of HIES appear to be prone to developing autoimmune diseases. We present two cases of HIES with autoimmune complications; one case was a product of a consanguineous marriage, the other one was a sporadic case. The first patient presented with recurrent episodes of erythema nodosum, warts, bronchiolitis obliterans and thrombocytopenia. The second patient developed glomerulonephritis resulting in endstage renal failure. She later developed malar rash, oral ulcers, cerebral infarcts with vasculitis and positive ANA, anti-dsDNA, and antiphospholipid antibodies. We discuss the dilemma in treating patients who present with both primary immunodeficiency and autoimmunity.</div></front></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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