ChloroquineV1, France, Analysis, bibRecord, 000227

Granulomatous Disease in CVID: Retrospective Analysis of Clinical Characteristics and Treatment Efficacy in a Cohort of 59 Patients

Identifieur interne : 000227 ( France/Analysis ); précédent : 000226; suivant : 000228

Granulomatous Disease in CVID: Retrospective Analysis of Clinical Characteristics and Treatment Efficacy in a Cohort of 59 Patients

Auteurs : Jean-Nicolas Boursiquot [Canada] ; Laurence Gérard [France] ; Marion Malphettes [France] ; Claire Fieschi [France] ; Lionel Galicier [France] ; David Boutboul [France] ; Raphael Borie [France] ; Jean-François Viallard [France] ; Pauline Soulas-Sprauel [France] ; Alice Berezne [France] ; Arnaud Jaccard [France] ; Eric Hachulla [France] ; Julien Haroche [France] ; Nicolas Schleinitz [France] ; Laurent Têtu [France] ; Eric Oksenhendler [France]

Source :

Journal of Clinical Immunology [ 0271-9142 ] ; 2013-01-01.

RBID : ISTEX:6D0EC3A09AF8C287E72FDBC64FA5B17ADECD676B

English descriptors

KwdEn :
- CVID, antibody deficiency, granulomatous disease, primary immune deficiency, treatment.

Abstract

Abstract: Background: Granulomatous disease (GD) will develop in a subset of patients with common variable immunodeficiency (CVID). Little is known about the efficacy of therapeutic agents used for treating this disorder. Objective: To evaluate the efficacy of immunosuppressive drugs with the help of a set of clinical, biological and radiological criteria. Method: Clinical and laboratory features of CVID patients were collected from the French DEFI cohort, a prospective study on adults with hypogammaglobulinemia. The medical charts of 55 patients (93 %) of the GD cohort were reviewed. Results: Among 436 subjects with CVID, 59 patients (13.5 %) were diagnosed with GD. Of the 55 patients in whom medical charts were available, 32 patients received treatment for the granulomatous disease. Corticosteroids were the most frequently used drug. Complete response to treatment was infrequent. It was achieved with corticosteroids, cyclophosphamide, hydroxychloroquine, rituximab and methotrexate. Azathioprine, cyclosporine, mycophenolate mofetil, sirolimus, infliximab and thalidomide led to partial or absence of response. Complete and partial responses were observed in lymph nodes, lungs, liver, skin, bone marrow and central nervous system. Absent of response for gastrointestinal tract granulomas was noted in all cases of treatment attempt. Conclusion: CVID patients with GD exhibit a particular biological phenotype. Treatment should be considered in any symptomatic patient or if there is evidence of organ dysfunction. Corticosteroids are the drug of choice in most instances but response to treatment is often unsatisfactory.

Url:

DOI: 10.1007/s10875-012-9778-9

Affiliations:

Links to Exploration step

ISTEX:6D0EC3A09AF8C287E72FDBC64FA5B17ADECD676B

Le document en format XML

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<sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">Granulomatous Disease in CVID: Retrospective Analysis of Clinical Characteristics and Treatment Efficacy in a Cohort of 59 Patients</title>
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<author><name sortKey="Boutboul, David" sort="Boutboul, David" uniqKey="Boutboul D" first="David" last="Boutboul">David Boutboul</name>
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<wicri:regionArea>Department of Respirology, Hôpital Tenon, Paris</wicri:regionArea>
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<author><name sortKey="Viallard, Jean Francois" sort="Viallard, Jean Francois" uniqKey="Viallard J" first="Jean-François" last="Viallard">Jean-François Viallard</name>
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<wicri:noRegion>Pessac</wicri:noRegion>
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<author><name sortKey="Soulas Sprauel, Pauline" sort="Soulas Sprauel, Pauline" uniqKey="Soulas Sprauel P" first="Pauline" last="Soulas-Sprauel">Pauline Soulas-Sprauel</name>
<affiliation wicri:level="3"><country xml:lang="fr">France</country>
<wicri:regionArea>Department of Internal Medicine and INSERM UMR S737, Strasbourg</wicri:regionArea>
<placeName><region type="region">Grand Est</region>
<region type="old region">Alsace (région administrative)</region>
<settlement type="city">Strasbourg</settlement>
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<author><name sortKey="Berezne, Alice" sort="Berezne, Alice" uniqKey="Berezne A" first="Alice" last="Berezne">Alice Berezne</name>
<affiliation wicri:level="3"><country xml:lang="fr">France</country>
<wicri:regionArea>Department of Internal Medicine, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris, Paris</wicri:regionArea>
<placeName><region type="region">Île-de-France</region>
<region type="old region">Île-de-France</region>
<settlement type="city">Paris</settlement>
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<author><name sortKey="Jaccard, Arnaud" sort="Jaccard, Arnaud" uniqKey="Jaccard A" first="Arnaud" last="Jaccard">Arnaud Jaccard</name>
<affiliation wicri:level="3"><country xml:lang="fr">France</country>
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<author><name sortKey="Hachulla, Eric" sort="Hachulla, Eric" uniqKey="Hachulla E" first="Eric" last="Hachulla">Eric Hachulla</name>
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<author><name sortKey="Haroche, Julien" sort="Haroche, Julien" uniqKey="Haroche J" first="Julien" last="Haroche">Julien Haroche</name>
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<author><name sortKey="Tetu, Laurent" sort="Tetu, Laurent" uniqKey="Tetu L" first="Laurent" last="Têtu">Laurent Têtu</name>
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<author><name sortKey="Oksenhendler, Eric" sort="Oksenhendler, Eric" uniqKey="Oksenhendler E" first="Eric" last="Oksenhendler">Eric Oksenhendler</name>
<affiliation wicri:level="3"><country xml:lang="fr">France</country>
<wicri:regionArea>Department of Clinical Immunology, Hôpital Saint-Louis, Assistance Publique- Hôpitaux de Paris, 1, rue Claude-Vellefaux, 75010, Paris</wicri:regionArea>
<placeName><region type="region" nuts="2">Île-de-France</region>
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<monogr></monogr>
<series><title level="j">Journal of Clinical Immunology</title>
<title level="j" type="abbrev">J Clin Immunol</title>
<idno type="ISSN">0271-9142</idno>
<idno type="eISSN">1573-2592</idno>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>CVID</term>
<term>antibody deficiency</term>
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<front><div type="abstract" xml:lang="en">Abstract: Background: Granulomatous disease (GD) will develop in a subset of patients with common variable immunodeficiency (CVID). Little is known about the efficacy of therapeutic agents used for treating this disorder. Objective: To evaluate the efficacy of immunosuppressive drugs with the help of a set of clinical, biological and radiological criteria. Method: Clinical and laboratory features of CVID patients were collected from the French DEFI cohort, a prospective study on adults with hypogammaglobulinemia. The medical charts of 55 patients (93 %) of the GD cohort were reviewed. Results: Among 436 subjects with CVID, 59 patients (13.5 %) were diagnosed with GD. Of the 55 patients in whom medical charts were available, 32 patients received treatment for the granulomatous disease. Corticosteroids were the most frequently used drug. Complete response to treatment was infrequent. It was achieved with corticosteroids, cyclophosphamide, hydroxychloroquine, rituximab and methotrexate. Azathioprine, cyclosporine, mycophenolate mofetil, sirolimus, infliximab and thalidomide led to partial or absence of response. Complete and partial responses were observed in lymph nodes, lungs, liver, skin, bone marrow and central nervous system. Absent of response for gastrointestinal tract granulomas was noted in all cases of treatment attempt. Conclusion: CVID patients with GD exhibit a particular biological phenotype. Treatment should be considered in any symptomatic patient or if there is evidence of organ dysfunction. Corticosteroids are the drug of choice in most instances but response to treatment is often unsatisfactory.</div>
</front>
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<tree><country name="Canada"><noRegion><name sortKey="Boursiquot, Jean Nicolas" sort="Boursiquot, Jean Nicolas" uniqKey="Boursiquot J" first="Jean-Nicolas" last="Boursiquot">Jean-Nicolas Boursiquot</name>
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<country name="France"><region name="Île-de-France"><name sortKey="Gerard, Laurence" sort="Gerard, Laurence" uniqKey="Gerard L" first="Laurence" last="Gérard">Laurence Gérard</name>
</region>
<name sortKey="Berezne, Alice" sort="Berezne, Alice" uniqKey="Berezne A" first="Alice" last="Berezne">Alice Berezne</name>
<name sortKey="Borie, Raphael" sort="Borie, Raphael" uniqKey="Borie R" first="Raphael" last="Borie">Raphael Borie</name>
<name sortKey="Boutboul, David" sort="Boutboul, David" uniqKey="Boutboul D" first="David" last="Boutboul">David Boutboul</name>
<name sortKey="Fieschi, Claire" sort="Fieschi, Claire" uniqKey="Fieschi C" first="Claire" last="Fieschi">Claire Fieschi</name>
<name sortKey="Galicier, Lionel" sort="Galicier, Lionel" uniqKey="Galicier L" first="Lionel" last="Galicier">Lionel Galicier</name>
<name sortKey="Hachulla, Eric" sort="Hachulla, Eric" uniqKey="Hachulla E" first="Eric" last="Hachulla">Eric Hachulla</name>
<name sortKey="Haroche, Julien" sort="Haroche, Julien" uniqKey="Haroche J" first="Julien" last="Haroche">Julien Haroche</name>
<name sortKey="Jaccard, Arnaud" sort="Jaccard, Arnaud" uniqKey="Jaccard A" first="Arnaud" last="Jaccard">Arnaud Jaccard</name>
<name sortKey="Malphettes, Marion" sort="Malphettes, Marion" uniqKey="Malphettes M" first="Marion" last="Malphettes">Marion Malphettes</name>
<name sortKey="Oksenhendler, Eric" sort="Oksenhendler, Eric" uniqKey="Oksenhendler E" first="Eric" last="Oksenhendler">Eric Oksenhendler</name>
<name sortKey="Schleinitz, Nicolas" sort="Schleinitz, Nicolas" uniqKey="Schleinitz N" first="Nicolas" last="Schleinitz">Nicolas Schleinitz</name>
<name sortKey="Soulas Sprauel, Pauline" sort="Soulas Sprauel, Pauline" uniqKey="Soulas Sprauel P" first="Pauline" last="Soulas-Sprauel">Pauline Soulas-Sprauel</name>
<name sortKey="Tetu, Laurent" sort="Tetu, Laurent" uniqKey="Tetu L" first="Laurent" last="Têtu">Laurent Têtu</name>
<name sortKey="Viallard, Jean Francois" sort="Viallard, Jean Francois" uniqKey="Viallard J" first="Jean-François" last="Viallard">Jean-François Viallard</name>
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Granulomatous Disease in CVID: Retrospective Analysis of Clinical Characteristics and Treatment Efficacy in a Cohort of 59 Patients

Granulomatous Disease in CVID: Retrospective Analysis of Clinical Characteristics and Treatment Efficacy in a Cohort of 59 Patients

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