Interstitial Lung Disease-Complicated Anti-MDA5 Antibody in Clinically Amyopathic Dermatomyositis Patients: Report of Two Cases With Distinct Clinical Features
Identifieur interne : 000006 ( France/Analysis ); précédent : 000005; suivant : 000007Interstitial Lung Disease-Complicated Anti-MDA5 Antibody in Clinically Amyopathic Dermatomyositis Patients: Report of Two Cases With Distinct Clinical Features
Auteurs : Laurence Pacot [France] ; Jacques Pouchot [France] ; Nicolas De Prost [France] ; Marie Senant [France] ; Eric Tartour [France] ; Françoise Le Pimpec-Barthes [France] ; Dominique Israel-Biet [France] ; Marie-Agnes Dragon-Durey [France]Source :
- Frontiers in Medicine [ 2296-858X ] ; 2020.
Abstract
Two patients presented simultaneously to our hospital with distinct clinical features associated with the presence of anti-MDA5 antibodies: the first one was admitted for a skin rash resembling to a toxic epidermal necrosis (Lyell syndrome) and the second one presented with pulmonary manifestations attributed to a diffuse fibrosing interstitial pneumonitis on chest CT-scan. In addition to the skin lesions involving 40% of the body surface area, the first patient developed a rapid diffuse interstitial pneumonitis with respiratory distress justifying the initiation of a systemic immunosuppressive treatment. However, she died 3 weeks after her admission from mesenteric thrombosis associated with septic shock. The second patient respiratory condition worsened despite an intensive immunosuppressive treatment with high doses of intravenous methylprednisolone and cyclophosphamide and plasmapheresis, and required lung transplantation. Anti-MDA5 antibody titer declined and disappeared on anti-rejection treatment. These two cases underline the diagnostic conundrum and the therapeutic difficulties in patients with anti-MDA5 antibodies and clinically amyopathic dermatomyositis (CADM) or interstitial lung disease (ILD), who may undergo rapidly-progressive and fatal outcome. Presence of anti-MDA5 antibodies should always be suspected when confronted to CADM patients with cutaneous ulcerations or ILD to allow a rapid and adapted treatment initiation.
Url:
DOI: 10.3389/fmed.2020.00077
PubMed: NONE
PubMed Central: 7076087
Affiliations:
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strict</wicri:regionArea></affiliation></author><author><name sortKey="Dragon Durey, Marie Agnes" sort="Dragon Durey, Marie Agnes" uniqKey="Dragon Durey M" first="Marie-Agnes" last="Dragon-Durey">Marie-Agnes Dragon-Durey</name><affiliation wicri:level="1"><nlm:aff id="aff1"><institution>Service d'Immunologie Biologique, Hôpital Européen Georges Pompidou</institution>,<addr-line>Paris</addr-line>,<country>France</country></nlm:aff><country xml:lang="fr">France</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation><affiliation wicri:level="1"><nlm:aff id="aff4"><institution>Université Paris-Descartes</institution>,<addr-line>Paris</addr-line>,<country>France</country></nlm:aff><country xml:lang="fr">France</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author></analytic><series><title level="j">Frontiers in Medicine</title><idno type="eISSN">2296-858X</idno><imprint><date when="2020">2020</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Two patients presented simultaneously to our hospital with distinct clinical features associated with the presence of anti-MDA5 antibodies: the first one was admitted for a skin rash resembling to a toxic epidermal necrosis (Lyell syndrome) and the second one presented with pulmonary manifestations attributed to a diffuse fibrosing interstitial pneumonitis on chest CT-scan. In addition to the skin lesions involving 40% of the body surface area, the first patient developed a rapid diffuse interstitial pneumonitis with respiratory distress justifying the initiation of a systemic immunosuppressive treatment. However, she died 3 weeks after her admission from mesenteric thrombosis associated with septic shock. The second patient respiratory condition worsened despite an intensive immunosuppressive treatment with high doses of intravenous methylprednisolone and cyclophosphamide and plasmapheresis, and required lung transplantation. Anti-MDA5 antibody titer declined and disappeared on anti-rejection treatment. These two cases underline the diagnostic conundrum and the therapeutic difficulties in patients with anti-MDA5 antibodies and clinically amyopathic dermatomyositis (CADM) or interstitial lung disease (ILD), who may undergo rapidly-progressive and fatal outcome. Presence of anti-MDA5 antibodies should always be suspected when confronted to CADM patients with cutaneous ulcerations or ILD to allow a rapid and adapted treatment initiation.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Fathi, M" uniqKey="Fathi M">M Fathi</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ortiz Santamaria, V" uniqKey="Ortiz Santamaria V">V Ortiz-Santamaria</name></author><author><name sortKey="Babot, A" uniqKey="Babot A">A Babot</name></author><author><name sortKey="Ferrer, C" uniqKey="Ferrer C">C Ferrer</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Arboleda, R" uniqKey="Arboleda R">R Arboleda</name></author><author><name sortKey="Gonzalez, O" uniqKey="Gonzalez O">O Gonzalez</name></author><author><name sortKey="Cortes, M" uniqKey="Cortes M">M Cortes</name></author><author><name sortKey="Perez Cerda, F" uniqKey="Perez Cerda F">F 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uniqKey="Kawaguchi Y">Y Kawaguchi</name></author><author><name sortKey="Kuwana, M" uniqKey="Kuwana M">M Kuwana</name></author><author><name sortKey="Hanaoka, M" uniqKey="Hanaoka M">M Hanaoka</name></author><author><name sortKey="Katsumata, Y" uniqKey="Katsumata Y">Y Katsumata</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Matsushita, T" uniqKey="Matsushita T">T Matsushita</name></author><author><name sortKey="Mizumaki, K" uniqKey="Mizumaki K">K Mizumaki</name></author><author><name sortKey="Kano, M" uniqKey="Kano M">M Kano</name></author><author><name sortKey="Yagi, N" uniqKey="Yagi N">N Yagi</name></author><author><name sortKey="Tennichi, M" uniqKey="Tennichi M">M Tennichi</name></author><author><name sortKey="Takeuchi, A" uniqKey="Takeuchi A">A Takeuchi</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="So, H" uniqKey="So H">H So</name></author><author><name sortKey="Wong, Vtl" uniqKey="Wong V">VTL Wong</name></author><author><name sortKey="Lao, Vwn" uniqKey="Lao V">VWN Lao</name></author><author><name sortKey="Pang, Ht" uniqKey="Pang H">HT Pang</name></author><author><name sortKey="Yip, Rml" uniqKey="Yip R">RML Yip</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Abe, Y" uniqKey="Abe Y">Y Abe</name></author><author><name sortKey="Kusaoi, M" uniqKey="Kusaoi M">M Kusaoi</name></author><author><name sortKey="Tada, K" uniqKey="Tada K">K Tada</name></author><author><name sortKey="Yamaji, K" uniqKey="Yamaji K">K Yamaji</name></author><author><name sortKey="Tamura, N" uniqKey="Tamura N">N Tamura</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hoa, S" uniqKey="Hoa S">S Hoa</name></author><author><name sortKey="Troyanov, Y" uniqKey="Troyanov Y">Y Troyanov</name></author><author><name sortKey="Fritzler, Mj" uniqKey="Fritzler M">MJ Fritzler</name></author><author><name sortKey="Targoff, In" uniqKey="Targoff I">IN Targoff</name></author><author><name sortKey="Chartrand, S" uniqKey="Chartrand S">S Chartrand</name></author><author><name sortKey="Mansour, Am" uniqKey="Mansour A">AM Mansour</name></author></analytic></biblStruct></listBibl></div1></back></TEI><affiliations><list><country><li>France</li></country></list><tree><country name="France"><noRegion><name sortKey="Pacot, Laurence" sort="Pacot, Laurence" uniqKey="Pacot L" first="Laurence" last="Pacot">Laurence Pacot</name></noRegion><name sortKey="De Prost, Nicolas" sort="De Prost, Nicolas" uniqKey="De Prost N" first="Nicolas" last="De Prost">Nicolas De Prost</name><name sortKey="Dragon Durey, Marie Agnes" sort="Dragon Durey, Marie Agnes" uniqKey="Dragon Durey M" first="Marie-Agnes" last="Dragon-Durey">Marie-Agnes Dragon-Durey</name><name sortKey="Dragon Durey, Marie Agnes" sort="Dragon Durey, Marie Agnes" uniqKey="Dragon Durey M" first="Marie-Agnes" last="Dragon-Durey">Marie-Agnes Dragon-Durey</name><name sortKey="Israel Biet, Dominique" sort="Israel Biet, Dominique" uniqKey="Israel Biet D" first="Dominique" last="Israel-Biet">Dominique Israel-Biet</name><name sortKey="Israel Biet, Dominique" sort="Israel Biet, Dominique" uniqKey="Israel Biet D" first="Dominique" last="Israel-Biet">Dominique Israel-Biet</name><name sortKey="Le Pimpec Barthes, Francoise" sort="Le Pimpec Barthes, Francoise" uniqKey="Le Pimpec Barthes F" first="Françoise" last="Le Pimpec-Barthes">Françoise Le Pimpec-Barthes</name><name sortKey="Pouchot, Jacques" sort="Pouchot, Jacques" uniqKey="Pouchot J" first="Jacques" last="Pouchot">Jacques Pouchot</name><name sortKey="Senant, Marie" sort="Senant, Marie" uniqKey="Senant M" first="Marie" last="Senant">Marie Senant</name><name sortKey="Senant, Marie" sort="Senant, Marie" uniqKey="Senant M" first="Marie" last="Senant">Marie Senant</name><name sortKey="Tartour, Eric" sort="Tartour, Eric" uniqKey="Tartour E" first="Eric" last="Tartour">Eric Tartour</name><name sortKey="Tartour, Eric" sort="Tartour, Eric" uniqKey="Tartour E" first="Eric" last="Tartour">Eric Tartour</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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