Macrophage activation syndrome in adult dermatomyositis: a case-based review
Macrophage activation syndrome (MAS) is a severe and life-threatening syndrome associated with autoimmune diseases, characterized by fever, hepatosplenomegaly, and pancytopenia. Dermatomyositis (DM) is one of the causes of MAS; however, its clinical characteristics in DM patients remain unclear. Thi...
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| Veröffentlicht in: | Rheumatology international 2020-07, Vol.40 (7), p.1151-1162 |
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| creator | Kishida, Dai Sakaguchi, Noriko Ueno, Ken-ichi Ushiyama, Satoru Ichikawa, Takanori Yoshinaga, Tsuneaki Shimojima, Yasuhiro Sekijima, Yoshiki |
| description | Macrophage activation syndrome (MAS) is a severe and life-threatening syndrome associated with autoimmune diseases, characterized by fever, hepatosplenomegaly, and pancytopenia. Dermatomyositis (DM) is one of the causes of MAS; however, its clinical characteristics in DM patients remain unclear. This study aimed to present a case of anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive DM complicated by MAS in a 29-year-old woman and to review the literatures including similar cases. Even though symptoms and cytopenia of our patient were refractory to combination therapy, including glucocorticoids, immunosuppressants, and plasma exchange, the administration of rituximab (RTX) resulted in rapid clinical improvement and glucocorticoid reduction. The literature review revealed 18 adult patients with DM associated MAS. Most patients developed MAS within 3 months from DM onset. A monotherapy of glucocorticoid was insufficient to control the disease, and the mortality of MAS in DM was higher than that of MAS in other rheumatic diseases, despite being treated by various means. RTX may be an effective treatment for patients with DM complicated by MAS who are refractory to conventional therapy. Anti-MDA5 antibody could influence the development of MAS; however, further investigations are needed to elucidate the association between myositis-specific antibody and MAS. |
| doi_str_mv | 10.1007/s00296-020-04590-9 |
| format | Article |
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Dermatomyositis (DM) is one of the causes of MAS; however, its clinical characteristics in DM patients remain unclear. This study aimed to present a case of anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive DM complicated by MAS in a 29-year-old woman and to review the literatures including similar cases. Even though symptoms and cytopenia of our patient were refractory to combination therapy, including glucocorticoids, immunosuppressants, and plasma exchange, the administration of rituximab (RTX) resulted in rapid clinical improvement and glucocorticoid reduction. The literature review revealed 18 adult patients with DM associated MAS. Most patients developed MAS within 3 months from DM onset. A monotherapy of glucocorticoid was insufficient to control the disease, and the mortality of MAS in DM was higher than that of MAS in other rheumatic diseases, despite being treated by various means. RTX may be an effective treatment for patients with DM complicated by MAS who are refractory to conventional therapy. Anti-MDA5 antibody could influence the development of MAS; however, further investigations are needed to elucidate the association between myositis-specific antibody and MAS.</description><identifier>ISSN: 0172-8172</identifier><identifier>EISSN: 1437-160X</identifier><identifier>DOI: 10.1007/s00296-020-04590-9</identifier><identifier>PMID: 32356114</identifier><language>eng</language><publisher>Berlin/Heidelberg: Springer Berlin Heidelberg</publisher><subject>Adult ; Antibodies ; Autoantibodies - immunology ; Case Based Review ; Dermatomyositis - complications ; Dermatomyositis - drug therapy ; Dermatomyositis - immunology ; Dermatomyositis - physiopathology ; Female ; Glucocorticoids - therapeutic use ; Humans ; Immunologic Factors - therapeutic use ; Immunosuppressive Agents - therapeutic use ; Immunotherapy ; Inflammatory diseases ; Interferon-Induced Helicase, IFIH1 - immunology ; Lung Diseases, Interstitial - complications ; Lung Diseases, Interstitial - diagnostic imaging ; Lung Diseases, Interstitial - drug therapy ; Lung Diseases, Interstitial - physiopathology ; Macrophage Activation Syndrome - drug therapy ; Macrophage Activation Syndrome - etiology ; Macrophage Activation Syndrome - immunology ; Medicine ; Medicine & Public Health ; Monoclonal antibodies ; Musculoskeletal diseases ; Plasma Exchange ; Rheumatology ; Rituximab - therapeutic use ; Targeted cancer therapy ; Tomography, X-Ray Computed</subject><ispartof>Rheumatology international, 2020-07, Vol.40 (7), p.1151-1162</ispartof><rights>Springer-Verlag GmbH Germany, part of Springer Nature 2020</rights><rights>Springer-Verlag GmbH Germany, part of Springer Nature 2020.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c403t-5425aa95124506b8d5687f8ad4a2ce5c72643c5f38ee5ba8595a0ffbf54abe623</citedby><cites>FETCH-LOGICAL-c403t-5425aa95124506b8d5687f8ad4a2ce5c72643c5f38ee5ba8595a0ffbf54abe623</cites><orcidid>0000-0001-7100-1121 ; 0000-0002-4323-0557 ; 0000-0001-8309-7984 ; 0000-0001-6570-5878 ; 0000-0002-8132-2743</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s00296-020-04590-9$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s00296-020-04590-9$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,780,784,27915,27916,41479,42548,51310</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32356114$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Kishida, Dai</creatorcontrib><creatorcontrib>Sakaguchi, Noriko</creatorcontrib><creatorcontrib>Ueno, Ken-ichi</creatorcontrib><creatorcontrib>Ushiyama, Satoru</creatorcontrib><creatorcontrib>Ichikawa, Takanori</creatorcontrib><creatorcontrib>Yoshinaga, Tsuneaki</creatorcontrib><creatorcontrib>Shimojima, Yasuhiro</creatorcontrib><creatorcontrib>Sekijima, Yoshiki</creatorcontrib><title>Macrophage activation syndrome in adult dermatomyositis: a case-based review</title><title>Rheumatology international</title><addtitle>Rheumatol Int</addtitle><addtitle>Rheumatol Int</addtitle><description>Macrophage activation syndrome (MAS) is a severe and life-threatening syndrome associated with autoimmune diseases, characterized by fever, hepatosplenomegaly, and pancytopenia. Dermatomyositis (DM) is one of the causes of MAS; however, its clinical characteristics in DM patients remain unclear. This study aimed to present a case of anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive DM complicated by MAS in a 29-year-old woman and to review the literatures including similar cases. Even though symptoms and cytopenia of our patient were refractory to combination therapy, including glucocorticoids, immunosuppressants, and plasma exchange, the administration of rituximab (RTX) resulted in rapid clinical improvement and glucocorticoid reduction. The literature review revealed 18 adult patients with DM associated MAS. Most patients developed MAS within 3 months from DM onset. A monotherapy of glucocorticoid was insufficient to control the disease, and the mortality of MAS in DM was higher than that of MAS in other rheumatic diseases, despite being treated by various means. RTX may be an effective treatment for patients with DM complicated by MAS who are refractory to conventional therapy. Anti-MDA5 antibody could influence the development of MAS; however, further investigations are needed to elucidate the association between myositis-specific antibody and MAS.</description><subject>Adult</subject><subject>Antibodies</subject><subject>Autoantibodies - immunology</subject><subject>Case Based Review</subject><subject>Dermatomyositis - complications</subject><subject>Dermatomyositis - drug therapy</subject><subject>Dermatomyositis - immunology</subject><subject>Dermatomyositis - physiopathology</subject><subject>Female</subject><subject>Glucocorticoids - therapeutic use</subject><subject>Humans</subject><subject>Immunologic Factors - therapeutic use</subject><subject>Immunosuppressive Agents - therapeutic use</subject><subject>Immunotherapy</subject><subject>Inflammatory diseases</subject><subject>Interferon-Induced Helicase, IFIH1 - immunology</subject><subject>Lung Diseases, Interstitial - complications</subject><subject>Lung Diseases, Interstitial - diagnostic imaging</subject><subject>Lung Diseases, Interstitial - drug therapy</subject><subject>Lung Diseases, Interstitial - physiopathology</subject><subject>Macrophage Activation Syndrome - drug therapy</subject><subject>Macrophage Activation Syndrome - etiology</subject><subject>Macrophage Activation Syndrome - immunology</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Monoclonal antibodies</subject><subject>Musculoskeletal diseases</subject><subject>Plasma Exchange</subject><subject>Rheumatology</subject><subject>Rituximab - therapeutic use</subject><subject>Targeted cancer therapy</subject><subject>Tomography, X-Ray Computed</subject><issn>0172-8172</issn><issn>1437-160X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNp9kU9P3DAQxa0KVBbaL9ADisSFi9vx_5gbWlFA2ooLSL1Zk2QCQZtkaydU--1rukClHrjMyPLPb57nMfZFwFcB4L4lAOktBwkctPHA_Qe2EFo5Liz83GMLEE7yMpcDdpjSI-SztfCRHSipjBVCL9jqB9Zx3DzgPRVYT90TTt04FGk7NHHsqeiGApt5PRUNxR6nsd-OqZu6dFZgUWMiXuXSFJGeOvr9ie23uE70-aUfsbvvF7fLK766ubxenq94rUFN3GhpEL0RUhuwVdkYW7q2xEajrMnUTlqtatOqkshUWBpvENq2ao3GiqxUR-x0p7uJ46-Z0hT6LtW0XuNA45yCVD5_VCjnM3ryH_o4znHI7oK0XjtdZhvvUhqcAeGVy5TcUXljKUVqwyZ2PcZtEBCeEwm7REJOJPxNJDwbOH6RnquemrcnrxFkQO2AlK-Ge4r_Zr8j-wfsqJUX</recordid><startdate>20200701</startdate><enddate>20200701</enddate><creator>Kishida, Dai</creator><creator>Sakaguchi, Noriko</creator><creator>Ueno, Ken-ichi</creator><creator>Ushiyama, Satoru</creator><creator>Ichikawa, Takanori</creator><creator>Yoshinaga, Tsuneaki</creator><creator>Shimojima, Yasuhiro</creator><creator>Sekijima, Yoshiki</creator><general>Springer Berlin Heidelberg</general><general>Springer Nature B.V</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0001-7100-1121</orcidid><orcidid>https://orcid.org/0000-0002-4323-0557</orcidid><orcidid>https://orcid.org/0000-0001-8309-7984</orcidid><orcidid>https://orcid.org/0000-0001-6570-5878</orcidid><orcidid>https://orcid.org/0000-0002-8132-2743</orcidid></search><sort><creationdate>20200701</creationdate><title>Macrophage activation syndrome in adult dermatomyositis: a case-based review</title><author>Kishida, Dai ; 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Dermatomyositis (DM) is one of the causes of MAS; however, its clinical characteristics in DM patients remain unclear. This study aimed to present a case of anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive DM complicated by MAS in a 29-year-old woman and to review the literatures including similar cases. Even though symptoms and cytopenia of our patient were refractory to combination therapy, including glucocorticoids, immunosuppressants, and plasma exchange, the administration of rituximab (RTX) resulted in rapid clinical improvement and glucocorticoid reduction. The literature review revealed 18 adult patients with DM associated MAS. Most patients developed MAS within 3 months from DM onset. A monotherapy of glucocorticoid was insufficient to control the disease, and the mortality of MAS in DM was higher than that of MAS in other rheumatic diseases, despite being treated by various means. RTX may be an effective treatment for patients with DM complicated by MAS who are refractory to conventional therapy. Anti-MDA5 antibody could influence the development of MAS; however, further investigations are needed to elucidate the association between myositis-specific antibody and MAS.</abstract><cop>Berlin/Heidelberg</cop><pub>Springer Berlin Heidelberg</pub><pmid>32356114</pmid><doi>10.1007/s00296-020-04590-9</doi><tpages>12</tpages><orcidid>https://orcid.org/0000-0001-7100-1121</orcidid><orcidid>https://orcid.org/0000-0002-4323-0557</orcidid><orcidid>https://orcid.org/0000-0001-8309-7984</orcidid><orcidid>https://orcid.org/0000-0001-6570-5878</orcidid><orcidid>https://orcid.org/0000-0002-8132-2743</orcidid></addata></record> |
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| subjects | Adult Antibodies Autoantibodies - immunology Case Based Review Dermatomyositis - complications Dermatomyositis - drug therapy Dermatomyositis - immunology Dermatomyositis - physiopathology Female Glucocorticoids - therapeutic use Humans Immunologic Factors - therapeutic use Immunosuppressive Agents - therapeutic use Immunotherapy Inflammatory diseases Interferon-Induced Helicase, IFIH1 - immunology Lung Diseases, Interstitial - complications Lung Diseases, Interstitial - diagnostic imaging Lung Diseases, Interstitial - drug therapy Lung Diseases, Interstitial - physiopathology Macrophage Activation Syndrome - drug therapy Macrophage Activation Syndrome - etiology Macrophage Activation Syndrome - immunology Medicine Medicine & Public Health Monoclonal antibodies Musculoskeletal diseases Plasma Exchange Rheumatology Rituximab - therapeutic use Targeted cancer therapy Tomography, X-Ray Computed |
| title | Macrophage activation syndrome in adult dermatomyositis: a case-based review |
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