Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis
Objective Autoantibodies recognizing specificity protein 4 (Sp4) were recently discovered in adults with idiopathic inflammatory myopathies (IIM). Anti‐Sp4 autoantibodies co‐occurred in patients with anti–transcription intermediary factor 1 (anti‐TIF1) autoantibody‐positive dermatomyositis (DM) and...
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| Veröffentlicht in: | Arthritis & rheumatology (Hoboken, N.J.) N.J.), 2023-09, Vol.75 (9), p.1668-1677 |
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| creator | Sherman, Matthew A. Pak, Katherine Pinal‐Fernandez, Iago Flegel, Willy A. Targoff, Ira N. Miller, Frederick W. Rider, Lisa G. Mammen, Andrew L. Albert, Daniel A. Arabshahi, Bita Balboni, Imelda M. Ballinger, Susan Bayat, Nastaran Bingham, C. April Bohnsack, John F. Cartwright, Victoria W. Cron, Randy Q. Curiel, Rodolfo Pena, Wendy Guzman, Marietta M. Eberhardt, Barbara Anne Edelheit, Barbara S. Farhadi, Payam Noroozi Finkel, Terri H. Fuhlbrigge, Robert C. Gewanter, Harry L. Goldmuntz, Ellen A. Gottlieb, Beth S. Griffin, Thomas A. Groh, Brandt P. Hannan, William P. Hawkins‐Holt, Melissa Henrickson, Michael Higgins, Gloria C. Imundo, Lisa Jansen, Anna Jarvis, James Jones, Olcay Y. Kamdar, Ankur Kim, Hanna Kingsbury, Daniel J. Kishi, Takayuki Lindsley, Carol B. Mamyrova, Gulnara McCarthy, Paul L. Mitchell, Stephen R. Nanda, Kabita Nativ, Simona Oral, Elif A. Pachman, Lauren M. Perez, Maria D. Person, Donald A. Rabinovich, Egla C. Ronis, Tova Sabbagh, Sara E. Sarkar, Kakali Schiffenbauer, Adam Shaham, Bracha Soep, Jennifer Stoll, Matthew L. Sule, Sangeeta Tarvin, Stacey E. Taylor‐Albert, Elizabeth Vogelgesang, Scott A. Volochayev, Rita White, Patience H. |
| description | Objective
Autoantibodies recognizing specificity protein 4 (Sp4) were recently discovered in adults with idiopathic inflammatory myopathies (IIM). Anti‐Sp4 autoantibodies co‐occurred in patients with anti–transcription intermediary factor 1 (anti‐TIF1) autoantibody‐positive dermatomyositis (DM) and were associated with a reduced risk of cancer. In the present study, the prevalence and clinical features associated with anti‐Sp4 autoantibodies in juvenile‐onset IIM were investigated.
Methods
Serum samples from 336 patients with juvenile myositis in a cross‐sectional cohort and 91 healthy controls were screened for anti‐Sp4 autoantibodies using enzyme‐linked immunosorbent assay. Clinical characteristics, outcomes, and HLA alleles of those with and those without anti‐Sp4 autoantibodies were compared.
Results
Anti‐Sp4 autoantibodies were present in 23 patients (7%) with juvenile myositis and were not present in any of the controls. Anti‐Sp4 autoantibodies were found among each clinical myositis subgroup. The frequency of TIF1 autoantibody positivity was significantly higher among those with anti‐Sp4 autoantibodies (21 [91%] versus 92 [30%], P |
| doi_str_mv | 10.1002/art.42512 |
| format | Article |
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Autoantibodies recognizing specificity protein 4 (Sp4) were recently discovered in adults with idiopathic inflammatory myopathies (IIM). Anti‐Sp4 autoantibodies co‐occurred in patients with anti–transcription intermediary factor 1 (anti‐TIF1) autoantibody‐positive dermatomyositis (DM) and were associated with a reduced risk of cancer. In the present study, the prevalence and clinical features associated with anti‐Sp4 autoantibodies in juvenile‐onset IIM were investigated.
Methods
Serum samples from 336 patients with juvenile myositis in a cross‐sectional cohort and 91 healthy controls were screened for anti‐Sp4 autoantibodies using enzyme‐linked immunosorbent assay. Clinical characteristics, outcomes, and HLA alleles of those with and those without anti‐Sp4 autoantibodies were compared.
Results
Anti‐Sp4 autoantibodies were present in 23 patients (7%) with juvenile myositis and were not present in any of the controls. Anti‐Sp4 autoantibodies were found among each clinical myositis subgroup. The frequency of TIF1 autoantibody positivity was significantly higher among those with anti‐Sp4 autoantibodies (21 [91%] versus 92 [30%], P < 0.001). In the anti‐TIF1 autoantibody–positive subgroup, Raynaud's phenomenon (8 [38%] versus 2 [2%], P < 0.001) was more common and peak aspartate aminotransferase was significantly lower in those with anti‐Sp4 autoantibodies. None of the patients with anti‐Sp4 autoantibodies required a wheelchair. Among White patients, DQA1*04 and DRB1*08 were associated with anti‐Sp4 autoantibodies.
Conclusion
Anti‐Sp4 autoantibodies were found in patients with juvenile‐onset IIM, predominantly those with coexisting anti‐TIF1 autoantibodies. Patients with anti‐Sp4 autoantibodies represent a phenotypic subset of anti‐TIF1 autoantibody–positive myositis characterized by frequent Raynaud's phenomenon and less pronounced muscle involvement, similar to adults with these autoantibodies. Novel immunogenetic risk factors for White patients with IIM were identified among juveniles with anti‐Sp4 autoantibodies.</description><identifier>ISSN: 2326-5191</identifier><identifier>ISSN: 2326-5205</identifier><identifier>EISSN: 2326-5205</identifier><identifier>DOI: 10.1002/art.42512</identifier><identifier>PMID: 36996276</identifier><language>eng</language><publisher>Boston, USA: Wiley Periodicals, Inc</publisher><subject>Adult ; Adults ; Aspartate aminotransferase ; Autoantibodies ; Cross-Sectional Studies ; Dermatomyositis ; DQA1 protein ; Drb1 protein ; Health risks ; Histocompatibility antigen HLA ; Humans ; Immunogenetics ; Inflammation ; Inflammatory diseases ; Juveniles ; Mediation Analysis ; Musculoskeletal diseases ; Myositis ; Proteins ; Raynaud disease ; Risk Factors ; Risk management ; Risk reduction ; Subgroups ; Wheelchairs</subject><ispartof>Arthritis & rheumatology (Hoboken, N.J.), 2023-09, Vol.75 (9), p.1668-1677</ispartof><rights>2023 American College of Rheumatology. This article has been contributed to by U.S. Government employees and their work is in the public domain in the USA.</rights><rights>2023 American College of Rheumatology</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3882-ffee61fbbccbe0f812dca73f6d4344167c3f021d88884acf3224b6876178b8fe3</citedby><cites>FETCH-LOGICAL-c3882-ffee61fbbccbe0f812dca73f6d4344167c3f021d88884acf3224b6876178b8fe3</cites><orcidid>0000-0001-6338-9218 ; 0000-0002-1631-7198 ; 0000-0003-3732-3252 ; 0000-0002-6912-2458 ; 0000-0003-2831-9593 ; 0000-0002-5448-1538</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fart.42512$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fart.42512$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,776,780,1411,27901,27902,45550,45551</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36996276$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Sherman, Matthew A.</creatorcontrib><creatorcontrib>Pak, Katherine</creatorcontrib><creatorcontrib>Pinal‐Fernandez, Iago</creatorcontrib><creatorcontrib>Flegel, Willy A.</creatorcontrib><creatorcontrib>Targoff, Ira N.</creatorcontrib><creatorcontrib>Miller, Frederick W.</creatorcontrib><creatorcontrib>Rider, Lisa G.</creatorcontrib><creatorcontrib>Mammen, Andrew L.</creatorcontrib><creatorcontrib>Albert, Daniel A.</creatorcontrib><creatorcontrib>Arabshahi, Bita</creatorcontrib><creatorcontrib>Balboni, Imelda M.</creatorcontrib><creatorcontrib>Ballinger, Susan</creatorcontrib><creatorcontrib>Bayat, Nastaran</creatorcontrib><creatorcontrib>Bingham, C. April</creatorcontrib><creatorcontrib>Bohnsack, John F.</creatorcontrib><creatorcontrib>Cartwright, Victoria W.</creatorcontrib><creatorcontrib>Cron, Randy Q.</creatorcontrib><creatorcontrib>Curiel, Rodolfo</creatorcontrib><creatorcontrib>Pena, Wendy</creatorcontrib><creatorcontrib>Guzman, Marietta M.</creatorcontrib><creatorcontrib>Eberhardt, Barbara Anne</creatorcontrib><creatorcontrib>Edelheit, Barbara S.</creatorcontrib><creatorcontrib>Farhadi, Payam Noroozi</creatorcontrib><creatorcontrib>Finkel, Terri H.</creatorcontrib><creatorcontrib>Fuhlbrigge, Robert C.</creatorcontrib><creatorcontrib>Gewanter, Harry L.</creatorcontrib><creatorcontrib>Goldmuntz, Ellen A.</creatorcontrib><creatorcontrib>Gottlieb, Beth S.</creatorcontrib><creatorcontrib>Griffin, Thomas A.</creatorcontrib><creatorcontrib>Groh, Brandt P.</creatorcontrib><creatorcontrib>Hannan, William P.</creatorcontrib><creatorcontrib>Hawkins‐Holt, Melissa</creatorcontrib><creatorcontrib>Henrickson, Michael</creatorcontrib><creatorcontrib>Higgins, Gloria C.</creatorcontrib><creatorcontrib>Imundo, Lisa</creatorcontrib><creatorcontrib>Jansen, Anna</creatorcontrib><creatorcontrib>Jarvis, James</creatorcontrib><creatorcontrib>Jones, Olcay Y.</creatorcontrib><creatorcontrib>Kamdar, Ankur</creatorcontrib><creatorcontrib>Kim, Hanna</creatorcontrib><creatorcontrib>Kingsbury, Daniel J.</creatorcontrib><creatorcontrib>Kishi, Takayuki</creatorcontrib><creatorcontrib>Lindsley, Carol B.</creatorcontrib><creatorcontrib>Mamyrova, Gulnara</creatorcontrib><creatorcontrib>McCarthy, Paul L.</creatorcontrib><creatorcontrib>Mitchell, Stephen R.</creatorcontrib><creatorcontrib>Nanda, Kabita</creatorcontrib><creatorcontrib>Nativ, Simona</creatorcontrib><creatorcontrib>Oral, Elif A.</creatorcontrib><creatorcontrib>Pachman, Lauren M.</creatorcontrib><creatorcontrib>Perez, Maria D.</creatorcontrib><creatorcontrib>Person, Donald A.</creatorcontrib><creatorcontrib>Rabinovich, Egla C.</creatorcontrib><creatorcontrib>Ronis, Tova</creatorcontrib><creatorcontrib>Sabbagh, Sara E.</creatorcontrib><creatorcontrib>Sarkar, Kakali</creatorcontrib><creatorcontrib>Schiffenbauer, Adam</creatorcontrib><creatorcontrib>Shaham, Bracha</creatorcontrib><creatorcontrib>Soep, Jennifer</creatorcontrib><creatorcontrib>Stoll, Matthew L.</creatorcontrib><creatorcontrib>Sule, Sangeeta</creatorcontrib><creatorcontrib>Tarvin, Stacey E.</creatorcontrib><creatorcontrib>Taylor‐Albert, Elizabeth</creatorcontrib><creatorcontrib>Vogelgesang, Scott A.</creatorcontrib><creatorcontrib>Volochayev, Rita</creatorcontrib><creatorcontrib>White, Patience H.</creatorcontrib><creatorcontrib>Childhood Myositis Heterogeneity Collaborative Study Group</creatorcontrib><creatorcontrib>for the Childhood Myositis Heterogeneity Collaborative Study Group</creatorcontrib><title>Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis</title><title>Arthritis & rheumatology (Hoboken, N.J.)</title><addtitle>Arthritis Rheumatol</addtitle><description>Objective
Autoantibodies recognizing specificity protein 4 (Sp4) were recently discovered in adults with idiopathic inflammatory myopathies (IIM). Anti‐Sp4 autoantibodies co‐occurred in patients with anti–transcription intermediary factor 1 (anti‐TIF1) autoantibody‐positive dermatomyositis (DM) and were associated with a reduced risk of cancer. In the present study, the prevalence and clinical features associated with anti‐Sp4 autoantibodies in juvenile‐onset IIM were investigated.
Methods
Serum samples from 336 patients with juvenile myositis in a cross‐sectional cohort and 91 healthy controls were screened for anti‐Sp4 autoantibodies using enzyme‐linked immunosorbent assay. Clinical characteristics, outcomes, and HLA alleles of those with and those without anti‐Sp4 autoantibodies were compared.
Results
Anti‐Sp4 autoantibodies were present in 23 patients (7%) with juvenile myositis and were not present in any of the controls. Anti‐Sp4 autoantibodies were found among each clinical myositis subgroup. The frequency of TIF1 autoantibody positivity was significantly higher among those with anti‐Sp4 autoantibodies (21 [91%] versus 92 [30%], P < 0.001). In the anti‐TIF1 autoantibody–positive subgroup, Raynaud's phenomenon (8 [38%] versus 2 [2%], P < 0.001) was more common and peak aspartate aminotransferase was significantly lower in those with anti‐Sp4 autoantibodies. None of the patients with anti‐Sp4 autoantibodies required a wheelchair. Among White patients, DQA1*04 and DRB1*08 were associated with anti‐Sp4 autoantibodies.
Conclusion
Anti‐Sp4 autoantibodies were found in patients with juvenile‐onset IIM, predominantly those with coexisting anti‐TIF1 autoantibodies. Patients with anti‐Sp4 autoantibodies represent a phenotypic subset of anti‐TIF1 autoantibody–positive myositis characterized by frequent Raynaud's phenomenon and less pronounced muscle involvement, similar to adults with these autoantibodies. Novel immunogenetic risk factors for White patients with IIM were identified among juveniles with anti‐Sp4 autoantibodies.</description><subject>Adult</subject><subject>Adults</subject><subject>Aspartate aminotransferase</subject><subject>Autoantibodies</subject><subject>Cross-Sectional Studies</subject><subject>Dermatomyositis</subject><subject>DQA1 protein</subject><subject>Drb1 protein</subject><subject>Health risks</subject><subject>Histocompatibility antigen HLA</subject><subject>Humans</subject><subject>Immunogenetics</subject><subject>Inflammation</subject><subject>Inflammatory diseases</subject><subject>Juveniles</subject><subject>Mediation Analysis</subject><subject>Musculoskeletal diseases</subject><subject>Myositis</subject><subject>Proteins</subject><subject>Raynaud disease</subject><subject>Risk Factors</subject><subject>Risk management</subject><subject>Risk reduction</subject><subject>Subgroups</subject><subject>Wheelchairs</subject><issn>2326-5191</issn><issn>2326-5205</issn><issn>2326-5205</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp1kcFu1DAURSMEolXpgh9AltjAYlrbSRxnGQ0MDCoCDYNYRo7zPLySsae2AxpW_QQk_oxP6JfgkikLJN7meXHe9dW9WfaY0TNGKT9XPp4VvGT8XnbMcy5mJafl_bs3q9lRdhrCJU1TV1TQ8mF2lIu6FrwSx9mvZoxO2Yid6xECWYF2G4vf0W7Ihx1oNKgx7sl77yKgJQWZu5vrH07r0ZNPGD-TJh3fXP9ce2WD9riL6CxZ2gh-Cz0qvycLpaPzhBFle9J4IE0ITqOK0E8SLzBEtDqS-YAWtRrIAlQcffJze7LcbkfrNmAhoiYrDF8OkoEkR2_Gr2BxAPJ27wJGDI-yB0YNAU4P-yT7uHi5nr-eXbx7tZw3FzOdS8lnxgAIZrpO6w6okYz3WlW5EX2RFwUTlc4N5ayXaQqlTc550QlZCVbJThrIT7Jnk-7Ou6sRQmy3GDQMg7LgxtDyqs5rKXghE_r0H_TSjd4mdy2XpSzTf6xO1POJ0t6F4MG0O4_blGDLaHvbdZu6bv90ndgnB8WxSzn_Je-aTcD5BHxL2ez_r9Q2q_Uk-Rv84rkN</recordid><startdate>202309</startdate><enddate>202309</enddate><creator>Sherman, Matthew A.</creator><creator>Pak, Katherine</creator><creator>Pinal‐Fernandez, Iago</creator><creator>Flegel, Willy A.</creator><creator>Targoff, Ira N.</creator><creator>Miller, Frederick W.</creator><creator>Rider, Lisa G.</creator><creator>Mammen, Andrew L.</creator><creator>Albert, Daniel A.</creator><creator>Arabshahi, Bita</creator><creator>Balboni, Imelda M.</creator><creator>Ballinger, Susan</creator><creator>Bayat, Nastaran</creator><creator>Bingham, C. 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April ; Bohnsack, John F. ; Cartwright, Victoria W. ; Cron, Randy Q. ; Curiel, Rodolfo ; Pena, Wendy ; Guzman, Marietta M. ; Eberhardt, Barbara Anne ; Edelheit, Barbara S. ; Farhadi, Payam Noroozi ; Finkel, Terri H. ; Fuhlbrigge, Robert C. ; Gewanter, Harry L. ; Goldmuntz, Ellen A. ; Gottlieb, Beth S. ; Griffin, Thomas A. ; Groh, Brandt P. ; Hannan, William P. ; Hawkins‐Holt, Melissa ; Henrickson, Michael ; Higgins, Gloria C. ; Imundo, Lisa ; Jansen, Anna ; Jarvis, James ; Jones, Olcay Y. ; Kamdar, Ankur ; Kim, Hanna ; Kingsbury, Daniel J. ; Kishi, Takayuki ; Lindsley, Carol B. ; Mamyrova, Gulnara ; McCarthy, Paul L. ; Mitchell, Stephen R. ; Nanda, Kabita ; Nativ, Simona ; Oral, Elif A. ; Pachman, Lauren M. ; Perez, Maria D. ; Person, Donald A. ; Rabinovich, Egla C. ; Ronis, Tova ; Sabbagh, Sara E. ; Sarkar, Kakali ; Schiffenbauer, Adam ; Shaham, Bracha ; Soep, Jennifer ; Stoll, Matthew L. ; Sule, Sangeeta ; Tarvin, Stacey E. ; Taylor‐Albert, Elizabeth ; Vogelgesang, Scott A. ; Volochayev, Rita ; White, Patience H.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3882-ffee61fbbccbe0f812dca73f6d4344167c3f021d88884acf3224b6876178b8fe3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2023</creationdate><topic>Adult</topic><topic>Adults</topic><topic>Aspartate aminotransferase</topic><topic>Autoantibodies</topic><topic>Cross-Sectional Studies</topic><topic>Dermatomyositis</topic><topic>DQA1 protein</topic><topic>Drb1 protein</topic><topic>Health risks</topic><topic>Histocompatibility antigen HLA</topic><topic>Humans</topic><topic>Immunogenetics</topic><topic>Inflammation</topic><topic>Inflammatory diseases</topic><topic>Juveniles</topic><topic>Mediation Analysis</topic><topic>Musculoskeletal diseases</topic><topic>Myositis</topic><topic>Proteins</topic><topic>Raynaud disease</topic><topic>Risk Factors</topic><topic>Risk management</topic><topic>Risk reduction</topic><topic>Subgroups</topic><topic>Wheelchairs</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Sherman, Matthew A.</creatorcontrib><creatorcontrib>Pak, Katherine</creatorcontrib><creatorcontrib>Pinal‐Fernandez, Iago</creatorcontrib><creatorcontrib>Flegel, Willy A.</creatorcontrib><creatorcontrib>Targoff, Ira N.</creatorcontrib><creatorcontrib>Miller, Frederick W.</creatorcontrib><creatorcontrib>Rider, Lisa G.</creatorcontrib><creatorcontrib>Mammen, Andrew L.</creatorcontrib><creatorcontrib>Albert, Daniel A.</creatorcontrib><creatorcontrib>Arabshahi, Bita</creatorcontrib><creatorcontrib>Balboni, Imelda M.</creatorcontrib><creatorcontrib>Ballinger, Susan</creatorcontrib><creatorcontrib>Bayat, Nastaran</creatorcontrib><creatorcontrib>Bingham, C. April</creatorcontrib><creatorcontrib>Bohnsack, John F.</creatorcontrib><creatorcontrib>Cartwright, Victoria W.</creatorcontrib><creatorcontrib>Cron, Randy Q.</creatorcontrib><creatorcontrib>Curiel, Rodolfo</creatorcontrib><creatorcontrib>Pena, Wendy</creatorcontrib><creatorcontrib>Guzman, Marietta M.</creatorcontrib><creatorcontrib>Eberhardt, Barbara Anne</creatorcontrib><creatorcontrib>Edelheit, Barbara S.</creatorcontrib><creatorcontrib>Farhadi, Payam Noroozi</creatorcontrib><creatorcontrib>Finkel, Terri H.</creatorcontrib><creatorcontrib>Fuhlbrigge, Robert C.</creatorcontrib><creatorcontrib>Gewanter, Harry L.</creatorcontrib><creatorcontrib>Goldmuntz, Ellen A.</creatorcontrib><creatorcontrib>Gottlieb, Beth S.</creatorcontrib><creatorcontrib>Griffin, Thomas A.</creatorcontrib><creatorcontrib>Groh, Brandt P.</creatorcontrib><creatorcontrib>Hannan, William P.</creatorcontrib><creatorcontrib>Hawkins‐Holt, Melissa</creatorcontrib><creatorcontrib>Henrickson, Michael</creatorcontrib><creatorcontrib>Higgins, Gloria C.</creatorcontrib><creatorcontrib>Imundo, Lisa</creatorcontrib><creatorcontrib>Jansen, Anna</creatorcontrib><creatorcontrib>Jarvis, James</creatorcontrib><creatorcontrib>Jones, Olcay Y.</creatorcontrib><creatorcontrib>Kamdar, Ankur</creatorcontrib><creatorcontrib>Kim, Hanna</creatorcontrib><creatorcontrib>Kingsbury, Daniel J.</creatorcontrib><creatorcontrib>Kishi, Takayuki</creatorcontrib><creatorcontrib>Lindsley, Carol B.</creatorcontrib><creatorcontrib>Mamyrova, Gulnara</creatorcontrib><creatorcontrib>McCarthy, Paul L.</creatorcontrib><creatorcontrib>Mitchell, Stephen R.</creatorcontrib><creatorcontrib>Nanda, Kabita</creatorcontrib><creatorcontrib>Nativ, Simona</creatorcontrib><creatorcontrib>Oral, Elif A.</creatorcontrib><creatorcontrib>Pachman, Lauren M.</creatorcontrib><creatorcontrib>Perez, Maria D.</creatorcontrib><creatorcontrib>Person, Donald A.</creatorcontrib><creatorcontrib>Rabinovich, Egla C.</creatorcontrib><creatorcontrib>Ronis, Tova</creatorcontrib><creatorcontrib>Sabbagh, Sara E.</creatorcontrib><creatorcontrib>Sarkar, Kakali</creatorcontrib><creatorcontrib>Schiffenbauer, Adam</creatorcontrib><creatorcontrib>Shaham, Bracha</creatorcontrib><creatorcontrib>Soep, Jennifer</creatorcontrib><creatorcontrib>Stoll, Matthew L.</creatorcontrib><creatorcontrib>Sule, Sangeeta</creatorcontrib><creatorcontrib>Tarvin, Stacey E.</creatorcontrib><creatorcontrib>Taylor‐Albert, Elizabeth</creatorcontrib><creatorcontrib>Vogelgesang, Scott A.</creatorcontrib><creatorcontrib>Volochayev, Rita</creatorcontrib><creatorcontrib>White, Patience H.</creatorcontrib><creatorcontrib>Childhood Myositis Heterogeneity Collaborative Study Group</creatorcontrib><creatorcontrib>for the Childhood Myositis Heterogeneity Collaborative Study Group</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Bacteriology Abstracts (Microbiology B)</collection><collection>Calcium & Calcified Tissue Abstracts</collection><collection>Immunology Abstracts</collection><collection>Nucleic Acids Abstracts</collection><collection>Toxicology Abstracts</collection><collection>Environmental Sciences and Pollution Management</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>Arthritis & rheumatology (Hoboken, N.J.)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Sherman, Matthew A.</au><au>Pak, Katherine</au><au>Pinal‐Fernandez, Iago</au><au>Flegel, Willy A.</au><au>Targoff, Ira N.</au><au>Miller, Frederick W.</au><au>Rider, Lisa G.</au><au>Mammen, Andrew L.</au><au>Albert, Daniel A.</au><au>Arabshahi, Bita</au><au>Balboni, Imelda M.</au><au>Ballinger, Susan</au><au>Bayat, Nastaran</au><au>Bingham, C. April</au><au>Bohnsack, John F.</au><au>Cartwright, Victoria W.</au><au>Cron, Randy Q.</au><au>Curiel, Rodolfo</au><au>Pena, Wendy</au><au>Guzman, Marietta M.</au><au>Eberhardt, Barbara Anne</au><au>Edelheit, Barbara S.</au><au>Farhadi, Payam Noroozi</au><au>Finkel, Terri H.</au><au>Fuhlbrigge, Robert C.</au><au>Gewanter, Harry L.</au><au>Goldmuntz, Ellen A.</au><au>Gottlieb, Beth S.</au><au>Griffin, Thomas A.</au><au>Groh, Brandt P.</au><au>Hannan, William P.</au><au>Hawkins‐Holt, Melissa</au><au>Henrickson, Michael</au><au>Higgins, Gloria C.</au><au>Imundo, Lisa</au><au>Jansen, Anna</au><au>Jarvis, James</au><au>Jones, Olcay Y.</au><au>Kamdar, Ankur</au><au>Kim, Hanna</au><au>Kingsbury, Daniel J.</au><au>Kishi, Takayuki</au><au>Lindsley, Carol B.</au><au>Mamyrova, Gulnara</au><au>McCarthy, Paul L.</au><au>Mitchell, Stephen R.</au><au>Nanda, Kabita</au><au>Nativ, Simona</au><au>Oral, Elif A.</au><au>Pachman, Lauren M.</au><au>Perez, Maria D.</au><au>Person, Donald A.</au><au>Rabinovich, Egla C.</au><au>Ronis, Tova</au><au>Sabbagh, Sara E.</au><au>Sarkar, Kakali</au><au>Schiffenbauer, Adam</au><au>Shaham, Bracha</au><au>Soep, Jennifer</au><au>Stoll, Matthew L.</au><au>Sule, Sangeeta</au><au>Tarvin, Stacey E.</au><au>Taylor‐Albert, Elizabeth</au><au>Vogelgesang, Scott A.</au><au>Volochayev, Rita</au><au>White, Patience H.</au><aucorp>Childhood Myositis Heterogeneity Collaborative Study Group</aucorp><aucorp>for the Childhood Myositis Heterogeneity Collaborative Study Group</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis</atitle><jtitle>Arthritis & rheumatology (Hoboken, N.J.)</jtitle><addtitle>Arthritis Rheumatol</addtitle><date>2023-09</date><risdate>2023</risdate><volume>75</volume><issue>9</issue><spage>1668</spage><epage>1677</epage><pages>1668-1677</pages><issn>2326-5191</issn><issn>2326-5205</issn><eissn>2326-5205</eissn><abstract>Objective
Autoantibodies recognizing specificity protein 4 (Sp4) were recently discovered in adults with idiopathic inflammatory myopathies (IIM). Anti‐Sp4 autoantibodies co‐occurred in patients with anti–transcription intermediary factor 1 (anti‐TIF1) autoantibody‐positive dermatomyositis (DM) and were associated with a reduced risk of cancer. In the present study, the prevalence and clinical features associated with anti‐Sp4 autoantibodies in juvenile‐onset IIM were investigated.
Methods
Serum samples from 336 patients with juvenile myositis in a cross‐sectional cohort and 91 healthy controls were screened for anti‐Sp4 autoantibodies using enzyme‐linked immunosorbent assay. Clinical characteristics, outcomes, and HLA alleles of those with and those without anti‐Sp4 autoantibodies were compared.
Results
Anti‐Sp4 autoantibodies were present in 23 patients (7%) with juvenile myositis and were not present in any of the controls. Anti‐Sp4 autoantibodies were found among each clinical myositis subgroup. The frequency of TIF1 autoantibody positivity was significantly higher among those with anti‐Sp4 autoantibodies (21 [91%] versus 92 [30%], P < 0.001). In the anti‐TIF1 autoantibody–positive subgroup, Raynaud's phenomenon (8 [38%] versus 2 [2%], P < 0.001) was more common and peak aspartate aminotransferase was significantly lower in those with anti‐Sp4 autoantibodies. None of the patients with anti‐Sp4 autoantibodies required a wheelchair. Among White patients, DQA1*04 and DRB1*08 were associated with anti‐Sp4 autoantibodies.
Conclusion
Anti‐Sp4 autoantibodies were found in patients with juvenile‐onset IIM, predominantly those with coexisting anti‐TIF1 autoantibodies. Patients with anti‐Sp4 autoantibodies represent a phenotypic subset of anti‐TIF1 autoantibody–positive myositis characterized by frequent Raynaud's phenomenon and less pronounced muscle involvement, similar to adults with these autoantibodies. Novel immunogenetic risk factors for White patients with IIM were identified among juveniles with anti‐Sp4 autoantibodies.</abstract><cop>Boston, USA</cop><pub>Wiley Periodicals, Inc</pub><pmid>36996276</pmid><doi>10.1002/art.42512</doi><tpages>10</tpages><orcidid>https://orcid.org/0000-0001-6338-9218</orcidid><orcidid>https://orcid.org/0000-0002-1631-7198</orcidid><orcidid>https://orcid.org/0000-0003-3732-3252</orcidid><orcidid>https://orcid.org/0000-0002-6912-2458</orcidid><orcidid>https://orcid.org/0000-0003-2831-9593</orcidid><orcidid>https://orcid.org/0000-0002-5448-1538</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 2326-5191 |
| ispartof | Arthritis & rheumatology (Hoboken, N.J.), 2023-09, Vol.75 (9), p.1668-1677 |
| issn | 2326-5191 2326-5205 2326-5205 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_2793986248 |
| source | MEDLINE; Wiley Online Library Journals Frontfile Complete; Alma/SFX Local Collection |
| subjects | Adult Adults Aspartate aminotransferase Autoantibodies Cross-Sectional Studies Dermatomyositis DQA1 protein Drb1 protein Health risks Histocompatibility antigen HLA Humans Immunogenetics Inflammation Inflammatory diseases Juveniles Mediation Analysis Musculoskeletal diseases Myositis Proteins Raynaud disease Risk Factors Risk management Risk reduction Subgroups Wheelchairs |
| title | Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis |
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