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Osseous sarcoidosis: a multicenter case-control study

Identifieur interne : 000020 ( France/Analysis ); précédent : 000019; suivant : 000021

Osseous sarcoidosis: a multicenter case-control study

Auteurs : Imen Ben Hassine [France] ; Christopher Rein [France] ; Cloé Comarmond [France] ; Camille Glanowski [France] ; Nathalie Saidenberg-Kermanac [France] ; Benoit Meunier [France] ; Nicolas Schleinitz [France] ; Noémie Chanson [France] ; Karim Sacre [France] ; Marc Scherlinger [France] ; Christophe Richez [France] ; Sandrine Hirschi [France] ; Matthieu Groh [France] ; Hervé Devilliers [France] ; Philip Bielefeld [France] ; David Saadoun [France] ; Catherine Chapelon-Abric [France] ; Laurent Arnaud [France] ; Patrice Cacoub [France]

Source :

RBID : Hal:hal-02291291

English descriptors

Abstract

Objective: To describe the clinical presentation, distribution of lesions, treatment, and outcomes of osseous sarcoidosis.Methods: A French retrospective multicenter study of patients with biopsy-proven sarcoidosis analyzed patients with 1) a biopsy-proven granuloma without caseous necrosis, and either 2) osseous clinical manifestations, or 3) abnormal osseous imaging. Sarcoidosis patients with osseous involvement (cases) were compared with 264 age- and sex-matched sarcoidosis patients with no osseous manifestations (controls).Results: In the osseous sarcoidosis group (n = 88), forty-two (48%) patients had osseous-related symptoms involving the axial (69%) and/or appendicular (58%) skeleton. On imaging, the most commonly affected bones were in the spine (52%), pelvis (42%), hands (22%) and femur (19%). Compared with controls, cases had higher rates of mediastinal (93% vs. 47%) and extra-thoracic lymph node involvement (66% vs. 21%), pulmonary (90% vs. 65%) and cutaneous involvement (44% vs. 23%) (all P < 0.0001), and hypercalcemia (8.5% vs. 2%, P = 0.014). Spleen/liver and gastrointestinal involvement were less frequent in the osseous sarcoidosis group (29% vs. 45%, and 1% vs. 17%, respectively, P < 0.0001). Response rates to with glucocorticoids alone, glucocorticoids plus methotrexate or glucocorticoids plus hydroxychloroquine were 23/44 (52%), 9/13 (69%) and 4/6 (67%), respectively.Conclusion: In patients with osseous sarcoidosis the spine and pelvis were the most commonly affected bones. Compared with controls, cases with osseous sarcoidosis have higher rates of thoracic and extra-thoracic lymph node involvement, pulmonary and cutaneous involvement, and hypercalcemia. Most patients with osseous sarcoidosis had a good response to glucocorticoids in combination with methotrexate or hydroxychloroquine.


Url:
DOI: 10.1016/j.jbspin.2019.07.009


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<hal:affiliation type="laboratory" xml:id="struct-475861" status="VALID">
<orgName>Service de médecine interne et maladies systémiques (SOC 2) [CHU de Dijon]</orgName>
<desc>
<address>
<addrLine>CHU de Dijon - 14 Rue Paul Gaffarel, 21000 Dijon</addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.chu-dijon.fr/fr/service/medecine-interne-maladies-systemiques-medecine-2-soc-2</ref>
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<org type="institution" xml:id="struct-300924" status="VALID">
<orgName>Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand</orgName>
<orgName type="acronym">CHU Dijon</orgName>
<desc>
<address>
<addrLine>CHU Dijon - 14 rue Paul Gaffarel - 21079 Dijon</addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.chu-dijon.fr/fr</ref>
</desc>
</org>
</tutelle>
</tutelles>
</hal:affiliation>
<country>France</country>
</affiliation>
</author>
<author>
<name sortKey="Saadoun, David" sort="Saadoun, David" uniqKey="Saadoun D" first="David" last="Saadoun">David Saadoun</name>
<affiliation wicri:level="1">
<hal:affiliation type="laboratory" xml:id="struct-542085" status="VALID">
<idno type="RNSR">200918530G</idno>
<orgName>Immunologie - Immunopathologie - Immunothérapie</orgName>
<orgName type="acronym">I3</orgName>
<desc>
<address>
<addrLine>Bâtiment CERVI, CHU Pitié-Salpêtrière 83 boulevard de l'Hôpital 75651 Paris Cedex 13 (France)</addrLine>
<country key="FR"></country>
</address>
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<org type="institution" xml:id="struct-303623" status="VALID">
<idno type="IdRef">026388278</idno>
<orgName>Institut National de la Santé et de la Recherche Médicale</orgName>
<orgName type="acronym">INSERM</orgName>
<desc>
<address>
<addrLine>101, rue de Tolbiac, 75013 Paris </addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.inserm.fr</ref>
</desc>
</org>
</tutelle>
<tutelle active="#struct-413221" type="direct">
<org type="institution" xml:id="struct-413221" status="VALID">
<orgName>Sorbonne Université</orgName>
<orgName type="acronym">SU</orgName>
<date type="start">2018-01-01</date>
<desc>
<address>
<addrLine>21 rue de l’École de médecine - 75006 Paris</addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.sorbonne-universite.fr/</ref>
</desc>
</org>
</tutelle>
<tutelle name="UMR7211" active="#struct-441569" type="direct">
<org type="institution" xml:id="struct-441569" status="VALID">
<idno type="IdRef">02636817X</idno>
<idno type="ISNI">0000000122597504</idno>
<orgName>Centre National de la Recherche Scientifique</orgName>
<orgName type="acronym">CNRS</orgName>
<date type="start">1939-10-19</date>
<desc>
<address>
<country key="FR"></country>
</address>
<ref type="url">http://www.cnrs.fr/</ref>
</desc>
</org>
</tutelle>
</tutelles>
</hal:affiliation>
<country>France</country>
</affiliation>
</author>
<author>
<name sortKey="Chapelon Abric, Catherine" sort="Chapelon Abric, Catherine" uniqKey="Chapelon Abric C" first="Catherine" last="Chapelon-Abric">Catherine Chapelon-Abric</name>
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<hal:affiliation type="department" xml:id="struct-454547" status="VALID">
<orgName>Service de médecine interne et d'immunologie clinique [CHU Pitié-Salpêtrière]</orgName>
<desc>
<address>
<country key="FR"></country>
</address>
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<org type="regroupinstitution" xml:id="struct-300068" status="VALID">
<orgName>Assistance publique - Hôpitaux de Paris (AP-HP)</orgName>
<orgName type="acronym">APHP</orgName>
<desc>
<address>
<country key="FR"></country>
</address>
</desc>
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<tutelle active="#struct-353778" type="direct">
<org type="institution" xml:id="struct-353778" status="VALID">
<orgName>CHU Pitié-Salpêtrière [APHP]</orgName>
<desc>
<address>
<addrLine>47-83 Boulevard de l'Hôpital, 75013 Paris</addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.aphp.fr/contenu/hopital-universitaire-pitie-salpetriere-0</ref>
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</org>
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</hal:affiliation>
<country>France</country>
</affiliation>
</author>
<author>
<name sortKey="Arnaud, Laurent" sort="Arnaud, Laurent" uniqKey="Arnaud L" first="Laurent" last="Arnaud">Laurent Arnaud</name>
<affiliation wicri:level="1">
<hal:affiliation type="laboratory" xml:id="struct-102530" status="VALID">
<orgName>Service de rhumatologie [Strasbourg]</orgName>
<desc>
<address>
<country key="FR"></country>
</address>
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<orgName>CHU Strasbourg</orgName>
<desc>
<address>
<country key="FR"></country>
</address>
</desc>
</org>
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<tutelle active="#struct-357391" type="direct">
<org type="institution" xml:id="struct-357391" status="VALID">
<orgName>Hôpital de Hautepierre [Strasbourg]</orgName>
<desc>
<address>
<addrLine>Avenue Molière67200 Strasbourg</addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.chru-strasbourg.fr/Hopital-de-Hautepierre</ref>
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</hal:affiliation>
<country>France</country>
</affiliation>
</author>
<author>
<name sortKey="Cacoub, Patrice" sort="Cacoub, Patrice" uniqKey="Cacoub P" first="Patrice" last="Cacoub">Patrice Cacoub</name>
<affiliation wicri:level="1">
<hal:affiliation type="department" xml:id="struct-454547" status="VALID">
<orgName>Service de médecine interne et d'immunologie clinique [CHU Pitié-Salpêtrière]</orgName>
<desc>
<address>
<country key="FR"></country>
</address>
</desc>
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<relation active="#struct-353778" type="direct"></relation>
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<tutelle active="#struct-300068" type="direct">
<org type="regroupinstitution" xml:id="struct-300068" status="VALID">
<orgName>Assistance publique - Hôpitaux de Paris (AP-HP)</orgName>
<orgName type="acronym">APHP</orgName>
<desc>
<address>
<country key="FR"></country>
</address>
</desc>
</org>
</tutelle>
<tutelle active="#struct-353778" type="direct">
<org type="institution" xml:id="struct-353778" status="VALID">
<orgName>CHU Pitié-Salpêtrière [APHP]</orgName>
<desc>
<address>
<addrLine>47-83 Boulevard de l'Hôpital, 75013 Paris</addrLine>
<country key="FR"></country>
</address>
<ref type="url">http://www.aphp.fr/contenu/hopital-universitaire-pitie-salpetriere-0</ref>
</desc>
</org>
</tutelle>
</tutelles>
</hal:affiliation>
<country>France</country>
</affiliation>
</author>
</analytic>
<idno type="DOI">10.1016/j.jbspin.2019.07.009</idno>
<series>
<title level="j">Joint Bone Spine</title>
<idno type="ISSN">1297-319X</idno>
<imprint>
<date type="datePub" subtype="inPress">2019</date>
</imprint>
</series>
</biblStruct>
</sourceDesc>
</fileDesc>
<profileDesc>
<textClass>
<keywords scheme="mix" xml:lang="en">
<term>Bone involvement</term>
<term>Osseous manifestations</term>
<term>Sarcoidosis</term>
</keywords>
</textClass>
</profileDesc>
</teiHeader>
<front>
<div type="abstract" xml:lang="en">
<p>Objective: To describe the clinical presentation, distribution of lesions, treatment, and outcomes of osseous sarcoidosis.Methods: A French retrospective multicenter study of patients with biopsy-proven sarcoidosis analyzed patients with 1) a biopsy-proven granuloma without caseous necrosis, and either 2) osseous clinical manifestations, or 3) abnormal osseous imaging. Sarcoidosis patients with osseous involvement (cases) were compared with 264 age- and sex-matched sarcoidosis patients with no osseous manifestations (controls).Results: In the osseous sarcoidosis group (n = 88), forty-two (48%) patients had osseous-related symptoms involving the axial (69%) and/or appendicular (58%) skeleton. On imaging, the most commonly affected bones were in the spine (52%), pelvis (42%), hands (22%) and femur (19%). Compared with controls, cases had higher rates of mediastinal (93% vs. 47%) and extra-thoracic lymph node involvement (66% vs. 21%), pulmonary (90% vs. 65%) and cutaneous involvement (44% vs. 23%) (all P < 0.0001), and hypercalcemia (8.5% vs. 2%, P = 0.014). Spleen/liver and gastrointestinal involvement were less frequent in the osseous sarcoidosis group (29% vs. 45%, and 1% vs. 17%, respectively, P < 0.0001). Response rates to with glucocorticoids alone, glucocorticoids plus methotrexate or glucocorticoids plus hydroxychloroquine were 23/44 (52%), 9/13 (69%) and 4/6 (67%), respectively.Conclusion: In patients with osseous sarcoidosis the spine and pelvis were the most commonly affected bones. Compared with controls, cases with osseous sarcoidosis have higher rates of thoracic and extra-thoracic lymph node involvement, pulmonary and cutaneous involvement, and hypercalcemia. Most patients with osseous sarcoidosis had a good response to glucocorticoids in combination with methotrexate or hydroxychloroquine.</p>
</div>
</front>
</TEI>
<affiliations>
<list>
<country>
<li>France</li>
</country>
<region>
<li>Provence-Alpes-Côte d'Azur</li>
<li>Île-de-France</li>
</region>
<settlement>
<li>Marseille</li>
<li>Paris</li>
</settlement>
<orgName>
<li>Université Paris 13</li>
<li>Université d'Aix-Marseille</li>
</orgName>
</list>
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<country name="France">
<noRegion>
<name sortKey="Ben Hassine, Imen" sort="Ben Hassine, Imen" uniqKey="Ben Hassine I" first="Imen" last="Ben Hassine">Imen Ben Hassine</name>
</noRegion>
<name sortKey="Arnaud, Laurent" sort="Arnaud, Laurent" uniqKey="Arnaud L" first="Laurent" last="Arnaud">Laurent Arnaud</name>
<name sortKey="Bielefeld, Philip" sort="Bielefeld, Philip" uniqKey="Bielefeld P" first="Philip" last="Bielefeld">Philip Bielefeld</name>
<name sortKey="Cacoub, Patrice" sort="Cacoub, Patrice" uniqKey="Cacoub P" first="Patrice" last="Cacoub">Patrice Cacoub</name>
<name sortKey="Chanson, Noemie" sort="Chanson, Noemie" uniqKey="Chanson N" first="Noémie" last="Chanson">Noémie Chanson</name>
<name sortKey="Chapelon Abric, Catherine" sort="Chapelon Abric, Catherine" uniqKey="Chapelon Abric C" first="Catherine" last="Chapelon-Abric">Catherine Chapelon-Abric</name>
<name sortKey="Comarmond, Cloe" sort="Comarmond, Cloe" uniqKey="Comarmond C" first="Cloé" last="Comarmond">Cloé Comarmond</name>
<name sortKey="Devilliers, Herve" sort="Devilliers, Herve" uniqKey="Devilliers H" first="Hervé" last="Devilliers">Hervé Devilliers</name>
<name sortKey="Glanowski, Camille" sort="Glanowski, Camille" uniqKey="Glanowski C" first="Camille" last="Glanowski">Camille Glanowski</name>
<name sortKey="Groh, Matthieu" sort="Groh, Matthieu" uniqKey="Groh M" first="Matthieu" last="Groh">Matthieu Groh</name>
<name sortKey="Hirschi, Sandrine" sort="Hirschi, Sandrine" uniqKey="Hirschi S" first="Sandrine" last="Hirschi">Sandrine Hirschi</name>
<name sortKey="Meunier, Benoit" sort="Meunier, Benoit" uniqKey="Meunier B" first="Benoit" last="Meunier">Benoit Meunier</name>
<name sortKey="Rein, Christopher" sort="Rein, Christopher" uniqKey="Rein C" first="Christopher" last="Rein">Christopher Rein</name>
<name sortKey="Richez, Christophe" sort="Richez, Christophe" uniqKey="Richez C" first="Christophe" last="Richez">Christophe Richez</name>
<name sortKey="Saadoun, David" sort="Saadoun, David" uniqKey="Saadoun D" first="David" last="Saadoun">David Saadoun</name>
<name sortKey="Sacre, Karim" sort="Sacre, Karim" uniqKey="Sacre K" first="Karim" last="Sacre">Karim Sacre</name>
<name sortKey="Saidenberg Kermanac, Nathalie" sort="Saidenberg Kermanac, Nathalie" uniqKey="Saidenberg Kermanac N" first="Nathalie" last="Saidenberg-Kermanac">Nathalie Saidenberg-Kermanac</name>
<name sortKey="Scherlinger, Marc" sort="Scherlinger, Marc" uniqKey="Scherlinger M" first="Marc" last="Scherlinger">Marc Scherlinger</name>
<name sortKey="Schleinitz, Nicolas" sort="Schleinitz, Nicolas" uniqKey="Schleinitz N" first="Nicolas" last="Schleinitz">Nicolas Schleinitz</name>
</country>
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</affiliations>
</record>

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