Association of anti-HSC70 autoantibodies with cutaneous ulceration and severe disease in juvenile dermatomyositis

Association of anti-HSC70 autoantibodies with cutaneous ulceration and severe disease in juvenile dermatomyositis

Abstract Objectives JDM is an inflammatory myopathy characterized by prominent vasculopathy. AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict o...

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Veröffentlicht in:Rheumatology (Oxford, England) England), 2022-07, Vol.61 (7), p.2969-2977
Hauptverfasser: Karasawa, Rie, Yudoh, Kazuo, Sato, Toshiko, Tanaka, Megumi, Tamaki, Mayumi, Sabbagh, Sara E, O’Hanlon, Terrance P, Noroozi-Farhadi, Payam, Targoff, Ira N, Flegel, Willy A, Mammen, Andrew L, Miller, Frederick W, Hicar, Mark D, Rider, Lisa G, Jarvis, James N
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container_end_page 2977
container_issue 7
container_start_page 2969
container_title Rheumatology (Oxford, England)
container_volume 61
creator Karasawa, Rie
Yudoh, Kazuo
Sato, Toshiko
Tanaka, Megumi
Tamaki, Mayumi
Sabbagh, Sara E
O’Hanlon, Terrance P
Noroozi-Farhadi, Payam
Targoff, Ira N
Flegel, Willy A
Mammen, Andrew L
Miller, Frederick W
Hicar, Mark D
Rider, Lisa G
Jarvis, James N
description Abstract Objectives JDM is an inflammatory myopathy characterized by prominent vasculopathy. AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict outcome. Methods Plasma samples from 63 patients with JDM, 49 patients with polyarticular JIA and 40 juvenile healthy controls were used to detect anti-heat shock cognate 71 kDa protein (HSC70) autoantibodies, a newly identified AECA, in ELISA assays. Clinical features were compared between JDM patients with and without anti-HSC70 autoantibodies. Results Anti-HSC70 autoantibodies were detected in 35% of patients with JDM, in 0% of patients with JIA (P 
doi_str_mv 10.1093/rheumatology/keab846
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AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict outcome. Methods Plasma samples from 63 patients with JDM, 49 patients with polyarticular JIA and 40 juvenile healthy controls were used to detect anti-heat shock cognate 71 kDa protein (HSC70) autoantibodies, a newly identified AECA, in ELISA assays. Clinical features were compared between JDM patients with and without anti-HSC70 autoantibodies. Results Anti-HSC70 autoantibodies were detected in 35% of patients with JDM, in 0% of patients with JIA (P &lt; 0.0001) and in 0% of healthy donors (P &lt; 0.0001). Both the presence of cutaneous ulcers (59% vs 17%, P &lt; 0.002) and the use of wheelchairs and/or assistive devices (64% vs 27%, P &lt; 0.007) were strongly associated with anti-HSC70 autoantibodies in JDM. High scores on the severity of myositis damage measures at the time of measurement of anti-HSC70 autoantibodies and an increased number of hospitalizations were also associated with anti-HSC70 autoantibodies. Intravenous immunoglobulin therapy was used more often in anti-HSC70 autoantibody-positive patients. Conclusion Anti-HCS70 autoantibodies are detected frequently in children with JDM and are novel myositis-associated autoantibodies correlating with disease severity.</description><identifier>ISSN: 1462-0324</identifier><identifier>EISSN: 1462-0332</identifier><identifier>DOI: 10.1093/rheumatology/keab846</identifier><identifier>PMID: 34791087</identifier><language>eng</language><publisher>Oxford University Press</publisher><subject>Clinical Science</subject><ispartof>Rheumatology (Oxford, England), 2022-07, Vol.61 (7), p.2969-2977</ispartof><rights>The Author(s) 2021. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For permissions, please email: journals.permissions@oup.com 2021</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c425t-3ad75e823973180b1aa05a832d57ee6a1dadd7c07ad778d79785b752b4fb34fc3</citedby><cites>FETCH-LOGICAL-c425t-3ad75e823973180b1aa05a832d57ee6a1dadd7c07ad778d79785b752b4fb34fc3</cites><orcidid>0000-0002-6912-2458 ; 0000-0003-3554-9194</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,776,780,881,1578,27901,27902</link.rule.ids></links><search><creatorcontrib>Karasawa, Rie</creatorcontrib><creatorcontrib>Yudoh, Kazuo</creatorcontrib><creatorcontrib>Sato, Toshiko</creatorcontrib><creatorcontrib>Tanaka, Megumi</creatorcontrib><creatorcontrib>Tamaki, Mayumi</creatorcontrib><creatorcontrib>Sabbagh, Sara E</creatorcontrib><creatorcontrib>O’Hanlon, Terrance P</creatorcontrib><creatorcontrib>Noroozi-Farhadi, Payam</creatorcontrib><creatorcontrib>Targoff, Ira N</creatorcontrib><creatorcontrib>Flegel, Willy A</creatorcontrib><creatorcontrib>Mammen, Andrew L</creatorcontrib><creatorcontrib>Miller, Frederick W</creatorcontrib><creatorcontrib>Hicar, Mark D</creatorcontrib><creatorcontrib>Rider, Lisa G</creatorcontrib><creatorcontrib>Jarvis, James N</creatorcontrib><title>Association of anti-HSC70 autoantibodies with cutaneous ulceration and severe disease in juvenile dermatomyositis</title><title>Rheumatology (Oxford, England)</title><description>Abstract Objectives JDM is an inflammatory myopathy characterized by prominent vasculopathy. AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict outcome. Methods Plasma samples from 63 patients with JDM, 49 patients with polyarticular JIA and 40 juvenile healthy controls were used to detect anti-heat shock cognate 71 kDa protein (HSC70) autoantibodies, a newly identified AECA, in ELISA assays. Clinical features were compared between JDM patients with and without anti-HSC70 autoantibodies. Results Anti-HSC70 autoantibodies were detected in 35% of patients with JDM, in 0% of patients with JIA (P &lt; 0.0001) and in 0% of healthy donors (P &lt; 0.0001). Both the presence of cutaneous ulcers (59% vs 17%, P &lt; 0.002) and the use of wheelchairs and/or assistive devices (64% vs 27%, P &lt; 0.007) were strongly associated with anti-HSC70 autoantibodies in JDM. High scores on the severity of myositis damage measures at the time of measurement of anti-HSC70 autoantibodies and an increased number of hospitalizations were also associated with anti-HSC70 autoantibodies. Intravenous immunoglobulin therapy was used more often in anti-HSC70 autoantibody-positive patients. Conclusion Anti-HCS70 autoantibodies are detected frequently in children with JDM and are novel myositis-associated autoantibodies correlating with disease severity.</description><subject>Clinical Science</subject><issn>1462-0324</issn><issn>1462-0332</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><recordid>eNqNkc9KAzEQxoMo_qm-gYe8wNpkkzTZi1CKWqHgQT0vs5vZNnW7qUm20re3pUX05mlmvpnfx8BHyC1nd5wVYhgW2K8g-dbPt8MPhMrI0Qm55HKUZ0yI_PSnz-UFuYpxyRhTXJhzciGkLjgz-pJ8jmP0tYPkfEd9Q6FLLpu-TjSj0Ce_HytvHUb65dKC1n2CDn0fad_WGA4YdJZG3GBAal1EiEhdR5f9BjvX7jQM-z9XWx9dcvGanDXQRrw51gF5f3x4m0yz2cvT82Q8y2qZq5QJsFqhyUWhBTes4gBMgRG5VRpxBNyCtbpmenenjdWFNqrSKq9kUwnZ1GJA7g--675aoa2xSwHach3cCsK29ODKv5vOLcq535RFroySYmcgDwZ18DEGbH5Yzsp9BOXvCMpjBDtseMB8v_4f8Q0LnJNX</recordid><startdate>20220706</startdate><enddate>20220706</enddate><creator>Karasawa, Rie</creator><creator>Yudoh, Kazuo</creator><creator>Sato, Toshiko</creator><creator>Tanaka, Megumi</creator><creator>Tamaki, Mayumi</creator><creator>Sabbagh, Sara E</creator><creator>O’Hanlon, Terrance P</creator><creator>Noroozi-Farhadi, Payam</creator><creator>Targoff, Ira N</creator><creator>Flegel, Willy A</creator><creator>Mammen, Andrew L</creator><creator>Miller, Frederick W</creator><creator>Hicar, Mark D</creator><creator>Rider, Lisa G</creator><creator>Jarvis, James N</creator><general>Oxford University Press</general><scope>AAYXX</scope><scope>CITATION</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-6912-2458</orcidid><orcidid>https://orcid.org/0000-0003-3554-9194</orcidid></search><sort><creationdate>20220706</creationdate><title>Association of anti-HSC70 autoantibodies with cutaneous ulceration and severe disease in juvenile dermatomyositis</title><author>Karasawa, Rie ; Yudoh, Kazuo ; Sato, Toshiko ; Tanaka, Megumi ; Tamaki, Mayumi ; Sabbagh, Sara E ; O’Hanlon, Terrance P ; Noroozi-Farhadi, Payam ; Targoff, Ira N ; Flegel, Willy A ; Mammen, Andrew L ; Miller, Frederick W ; Hicar, Mark D ; Rider, Lisa G ; Jarvis, James N</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c425t-3ad75e823973180b1aa05a832d57ee6a1dadd7c07ad778d79785b752b4fb34fc3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Clinical Science</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Karasawa, Rie</creatorcontrib><creatorcontrib>Yudoh, Kazuo</creatorcontrib><creatorcontrib>Sato, Toshiko</creatorcontrib><creatorcontrib>Tanaka, Megumi</creatorcontrib><creatorcontrib>Tamaki, Mayumi</creatorcontrib><creatorcontrib>Sabbagh, Sara E</creatorcontrib><creatorcontrib>O’Hanlon, Terrance P</creatorcontrib><creatorcontrib>Noroozi-Farhadi, Payam</creatorcontrib><creatorcontrib>Targoff, Ira N</creatorcontrib><creatorcontrib>Flegel, Willy A</creatorcontrib><creatorcontrib>Mammen, Andrew L</creatorcontrib><creatorcontrib>Miller, Frederick W</creatorcontrib><creatorcontrib>Hicar, Mark D</creatorcontrib><creatorcontrib>Rider, Lisa G</creatorcontrib><creatorcontrib>Jarvis, James N</creatorcontrib><collection>CrossRef</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Rheumatology (Oxford, England)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Karasawa, Rie</au><au>Yudoh, Kazuo</au><au>Sato, Toshiko</au><au>Tanaka, Megumi</au><au>Tamaki, Mayumi</au><au>Sabbagh, Sara E</au><au>O’Hanlon, Terrance P</au><au>Noroozi-Farhadi, Payam</au><au>Targoff, Ira N</au><au>Flegel, Willy A</au><au>Mammen, Andrew L</au><au>Miller, Frederick W</au><au>Hicar, Mark D</au><au>Rider, Lisa G</au><au>Jarvis, James N</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Association of anti-HSC70 autoantibodies with cutaneous ulceration and severe disease in juvenile dermatomyositis</atitle><jtitle>Rheumatology (Oxford, England)</jtitle><date>2022-07-06</date><risdate>2022</risdate><volume>61</volume><issue>7</issue><spage>2969</spage><epage>2977</epage><pages>2969-2977</pages><issn>1462-0324</issn><eissn>1462-0332</eissn><abstract>Abstract Objectives JDM is an inflammatory myopathy characterized by prominent vasculopathy. AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict outcome. Methods Plasma samples from 63 patients with JDM, 49 patients with polyarticular JIA and 40 juvenile healthy controls were used to detect anti-heat shock cognate 71 kDa protein (HSC70) autoantibodies, a newly identified AECA, in ELISA assays. Clinical features were compared between JDM patients with and without anti-HSC70 autoantibodies. Results Anti-HSC70 autoantibodies were detected in 35% of patients with JDM, in 0% of patients with JIA (P &lt; 0.0001) and in 0% of healthy donors (P &lt; 0.0001). Both the presence of cutaneous ulcers (59% vs 17%, P &lt; 0.002) and the use of wheelchairs and/or assistive devices (64% vs 27%, P &lt; 0.007) were strongly associated with anti-HSC70 autoantibodies in JDM. High scores on the severity of myositis damage measures at the time of measurement of anti-HSC70 autoantibodies and an increased number of hospitalizations were also associated with anti-HSC70 autoantibodies. Intravenous immunoglobulin therapy was used more often in anti-HSC70 autoantibody-positive patients. Conclusion Anti-HCS70 autoantibodies are detected frequently in children with JDM and are novel myositis-associated autoantibodies correlating with disease severity.</abstract><pub>Oxford University Press</pub><pmid>34791087</pmid><doi>10.1093/rheumatology/keab846</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0002-6912-2458</orcidid><orcidid>https://orcid.org/0000-0003-3554-9194</orcidid><oa>free_for_read</oa></addata></record>
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title Association of anti-HSC70 autoantibodies with cutaneous ulceration and severe disease in juvenile dermatomyositis
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