{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T06:16:03Z","timestamp":1790144163991,"version":"4.0.1"},"reference-count":52,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2023,3,7]],"date-time":"2023-03-07T00:00:00Z","timestamp":1678147200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100009619","name":"Japan Agency for Medical Research and Development","doi-asserted-by":"publisher","award":["JP21ek0109490h0002"],"award-info":[{"award-number":["JP21ek0109490h0002"]}],"id":[{"id":"10.13039\/100009619","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Brain Pathology"],"published-print":{"date-parts":[[2023,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>\n                    Identification of antisynthetase syndrome (ASS) could be challenging due to inaccessibility and technical difficulty of the serology test for the less common non\u2010Jo\u20101 antibodies. This study aimed to describe ASS antibody\u2010specific myopathology and evaluate the diagnostic utility of myofiber HLA\u2010DR expression. We reviewed 212 ASS muscle biopsies and compared myopathologic features among subtypes. Additionally, we compared their HLA\u2010DR staining pattern with 602 non\u2010ASS myositis and 140 genetically confirmed myopathies known to have an inflammatory component. We used\n                    <jats:italic>t<\/jats:italic>\n                    \u2010test and Fisher's exact for comparisons and used sensitivity, specificity, positive and negative predictive values to assess the utility of HLA\u2010DR expression for ASS diagnosis. RNAseq performed from a subset of myositis cases and histologically normal muscle biopsies was used to evaluate interferon (IFN)\u2010signaling pathway\u2010related genes. Anti\u2010OJ ASS showed prominent myopathology with higher scores in muscle fiber (4.6\u2009\u00b1\u20092.0 vs. 2.8\u2009\u00b1\u20091.8,\n                    <jats:italic>p<\/jats:italic>\n                    \u00a0=\u00a00.001) and inflammatory domains (6.8\u2009\u00b1\u20093.2 vs. 4.5\u2009\u00b1\u20092.9,\n                    <jats:italic>p<\/jats:italic>\n                    \u00a0=\u00a00.006) than non\u2010OJ ASS. HLA\u2010DR expression and IFN\u2010\u03b3\u2010related genes upregulation were prominent in ASS and inclusion body myositis (IBM). When dermatomyositis and IBM were excluded, HLA\u2010DR expression was 95.4% specific and 61.2% sensitive for ASS with a positive predictive value of 85.9% and a negative predictive value of 84.2%; perifascicular HLA\u2010DR pattern is common in anti\u2010Jo\u20101 ASS than non\u2010Jo\u20101 ASS (63.1% vs. 5.1%,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.0001). In the appropriate clinicopathological context, myofiber HLA\u2010DR expression help support ASS diagnosis. The presence of HLA\u2010DR expression suggests involvement of IFN\u2010\u03b3 in the pathogenesis of ASS, though the detailed mechanisms have yet to be elucidated.\n                  <\/jats:p>","DOI":"10.1111\/bpa.13155","type":"journal-article","created":{"date-parts":[[2023,3,7]],"date-time":"2023-03-07T18:29:40Z","timestamp":1678213780000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":42,"title":["Muscle pathology of antisynthetase syndrome according to antibody subtypes"],"prefix":"10.1111","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4110-9198","authenticated-orcid":false,"given":"Jantima","family":"Tanboon","sequence":"first","affiliation":[{"name":"Department of Neuromuscular Research, National Institute of Neuroscience National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"},{"name":"Department of Genome Medicine Development, Medical Genome Center National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michio","family":"Inoue","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Research, National Institute of Neuroscience National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"},{"name":"Department of Genome Medicine Development, Medical Genome Center National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinya","family":"Hirakawa","sequence":"additional","affiliation":[{"name":"Department of Clinical Data Science, Clinical Research &amp; Education Promotion Division National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hisateru","family":"Tachimori","sequence":"additional","affiliation":[{"name":"Department of Clinical Data Science, Clinical Research &amp; Education Promotion Division National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinichiro","family":"Hayashi","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Research, National Institute of Neuroscience National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"},{"name":"Department of Genome Medicine Development, Medical Genome Center National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoru","family":"Noguchi","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Research, National Institute of Neuroscience National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"},{"name":"Department of Genome Medicine Development, Medical Genome Center National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo 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Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9452-112X","authenticated-orcid":false,"given":"Ichizo","family":"Nishino","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Research, National Institute of Neuroscience National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"},{"name":"Department of Genome Medicine Development, Medical Genome Center National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo Japan"},{"name":"Department of Clinical Genome Analysis, Medical Genome Center National Center of Neurology and Psychiatry (NCNP)  Kodaira Tokyo 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