{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,22]],"date-time":"2026-04-22T13:58:12Z","timestamp":1776866292176,"version":"3.51.2"},"reference-count":27,"publisher":"Public Library of Science (PLoS)","issue":"4","license":[{"start":{"date-parts":[[2023,4,14]],"date-time":"2023-04-14T00:00:00Z","timestamp":1681430400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100002701","name":"Ministry of Education","doi-asserted-by":"publisher","award":["2020R1I1A1A01068066"],"award-info":[{"award-number":["2020R1I1A1A01068066"]}],"id":[{"id":"10.13039\/501100002701","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100008005","name":"Yonsei University College of Medicine","doi-asserted-by":"publisher","award":["6-2020-0127"],"award-info":[{"award-number":["6-2020-0127"]}],"id":[{"id":"10.13039\/501100008005","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:p>To explore the clinical significance of anti-cytosolic 5\u2019-nucleoditase 1A (NT5c1A) antibody seropositivity in inflammatory myopathies, we measured anti-NT5c1A antibodies and analyzed their clinical features. Anti-NT5c1A antibodies were measured in the sera of 103 patients with inflammatory myopathies using an enzyme-linked immunosorbent assay. Positivity for anti-NT5c1A antibody was found in 13 (12.6%) of 103 patients with inflammatory myopathy. Anti-NT5c1A antibody was most frequently identified in patients with inclusion body myositis (IBM) (8\/20, 40%), followed by dermatomyositis (2\/13, 15.4%), immune-mediated necrotizing myopathy (2\/28, 7.1%), and polymyositis (1\/42, 2.4%). In eight patients with the anti-NT5c1A antibody-seropositive IBM, the median age at symptom onset was 54 years (interquartile range [IQR]: 48\u201357 years), and the median disease duration was 34 months (IQR: 24\u201350 months]. Knee extension weakness was greater than or equal to hip flexion weakness in eight (100%) patients, and finger flexion strength was less than shoulder abduction in three (38%) patients. Dysphagia symptoms were found in three (38%) patients. The median serum CK level was 581 IU\/l (IQR: 434\u2013868 IU\/L]. Clinically significant differences were not found between anti-NT5c1A antibody-seropositive and seronegative IBM groups with respect to gender, age at symptom onset, age at diagnosis, disease duration, serum CK values, presence of other autoantibodies, dysphagia, and the pattern of muscle impairment. Although anti-NT5c1A antibody is known to be associated with IBM, seropositivity has also been noted in non-IBM inflammatory myopathies, and is insufficient to have clinical significance by itself. These findings have important implications for interpreting anti-NT5c1A antibody test results as the first study in Korea.<\/jats:p>","DOI":"10.1371\/journal.pone.0284409","type":"journal-article","created":{"date-parts":[[2023,4,14]],"date-time":"2023-04-14T17:31:28Z","timestamp":1681493488000},"page":"e0284409","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":7,"title":["Clinical significance of anti-NT5c1A autoantibody in Korean patients with inflammatory myopathies"],"prefix":"10.1371","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0727-638X","authenticated-orcid":true,"given":"Seung-Ah","family":"Lee","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hyun Joon","family":"Lee","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bum Chun","family":"Suh","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ha Young","family":"Shin","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Seung Woo","family":"Kim","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Byeol-A","family":"Yoon","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Young-Chul","family":"Choi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4165-8901","authenticated-orcid":true,"given":"Hyung Jun","family":"Park","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2023,4,14]]},"reference":[{"issue":"12","key":"pone.0284409.ref001","doi-asserted-by":"crossref","first-page":"1955","DOI":"10.1136\/annrheumdis-2017-211468","article-title":"2017 European League Against Rheumatism\/American College of Rheumatology classification criteria for adult and juvenile idiopathic inflammatory myopathies and their major subgroups","volume":"76","author":"IE Lundberg","year":"2017","journal-title":"Ann Rheum Dis"},{"issue":"1","key":"pone.0284409.ref002","doi-asserted-by":"crossref","first-page":"101484","DOI":"10.1016\/j.berh.2019.101484","article-title":"Inflammatory muscle disease\u2014An update","volume":"34","author":"S Baig","year":"2020","journal-title":"Best Pract Res Clin Rheumatol"},{"issue":"1","key":"pone.0284409.ref003","doi-asserted-by":"crossref","first-page":"86","DOI":"10.1038\/s41572-021-00321-x","article-title":"Idiopathic inflammatory myopathies","volume":"7","author":"IE Lundberg","year":"2021","journal-title":"Nat Rev Dis Primers"},{"issue":"1","key":"pone.0284409.ref004","doi-asserted-by":"crossref","first-page":"16","DOI":"10.1007\/s13317-016-0088-8","article-title":"Development and evaluation of a standardized ELISA for the determination of autoantibodies against cN-1A (Mup44, NT5C1A) in sporadic inclusion body myositis","volume":"7","author":"SL Kramp","year":"2016","journal-title":"Auto Immun Highlights"},{"issue":"8","key":"pone.0284409.ref005","doi-asserted-by":"crossref","first-page":"660","DOI":"10.1016\/j.nmd.2018.06.005","article-title":"Sensitivity and clinical utility of the anti-cytosolic 5\u2019-nucleotidase 1A (cN1A) antibody test in sporadic inclusion body myositis: Report of 40 patients from a single neuromuscular center","volume":"28","author":"KJ Felice","year":"2018","journal-title":"Neuromuscul Disord"},{"issue":"3","key":"pone.0284409.ref006","doi-asserted-by":"crossref","first-page":"397","DOI":"10.1002\/ana.23822","article-title":"Autoantibodies to cytosolic 5\u2019-nucleotidase 1A in inclusion body myositis","volume":"73","author":"H Pluk","year":"2013","journal-title":"Ann Neurol"},{"issue":"3","key":"pone.0284409.ref007","doi-asserted-by":"crossref","first-page":"408","DOI":"10.1002\/ana.23840","article-title":"Cytosolic 5\u2019-nucleotidase 1A autoimmunity in sporadic inclusion body myositis","volume":"73","author":"HB Larman","year":"2013","journal-title":"Ann Neurol"},{"issue":"4","key":"pone.0284409.ref008","doi-asserted-by":"crossref","first-page":"373","DOI":"10.1136\/jnnp-2014-310008","article-title":"Seropositivity for NT5c1A antibody in sporadic inclusion body myositis predicts more severe motor, bulbar and respiratory involvement","volume":"87","author":"NA Goyal","year":"2016","journal-title":"J Neurol Neurosurg Psychiatry"},{"issue":"4","key":"pone.0284409.ref009","doi-asserted-by":"crossref","first-page":"696","DOI":"10.1136\/annrheumdis-2014-206691","article-title":"Disease specificity of autoantibodies to cytosolic 5\u2019-nucleotidase 1A in sporadic inclusion body myositis versus known autoimmune diseases","volume":"75","author":"MK Herbert","year":"2016","journal-title":"Ann Rheum Dis"},{"issue":"1","key":"pone.0284409.ref010","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1002\/acr.22600","article-title":"Cytosolic 5\u2019-Nucleotidase 1A As a Target of Circulating Autoantibodies in Autoimmune Diseases","volume":"68","author":"TE Lloyd","year":"2016","journal-title":"Arthritis Care Res (Hoboken)"},{"issue":"4","key":"pone.0284409.ref011","doi-asserted-by":"crossref","first-page":"512","DOI":"10.1002\/ana.24919","article-title":"Pathomechanisms of anti-cytosolic 5\u2019-nucleotidase 1A autoantibodies in sporadic inclusion body myositis","volume":"81","author":"N Tawara","year":"2017","journal-title":"Ann Neurol"},{"issue":"5","key":"pone.0284409.ref012","doi-asserted-by":"crossref","first-page":"862","DOI":"10.1136\/annrheumdis-2016-210282","article-title":"Cytosolic 5\u2019-nucleotidase 1A autoantibody profile and clinical characteristics in inclusion body myositis","volume":"76","author":"JB Lilleker","year":"2017","journal-title":"Ann Rheum Dis"},{"issue":"3","key":"pone.0284409.ref013","doi-asserted-by":"crossref","first-page":"571","DOI":"10.1002\/acn3.51294","article-title":"Clinical utility of anti-cytosolic 5\u2019-nucleotidase 1A antibody in idiopathic inflammatory myopathies","volume":"8","author":"C Ikenaga","year":"2021","journal-title":"Ann Clin Transl Neurol"},{"issue":"1","key":"pone.0284409.ref014","doi-asserted-by":"crossref","first-page":"87","DOI":"10.1016\/j.nmd.2017.09.016","article-title":"224th ENMC International Workshop:: Clinico-sero-pathological classification of immune-mediated necrotizing myopathies Zandvoort, The Netherlands, 14\u201316 October 2016","volume":"28","author":"Y Allenbach","year":"2018","journal-title":"Neuromuscul Disord"},{"issue":"3","key":"pone.0284409.ref015","doi-asserted-by":"crossref","first-page":"403","DOI":"10.1111\/cge.12826","article-title":"Discovery of pathogenic variants in a large Korean cohort of inherited muscular disorders","volume":"91","author":"HJ Park","year":"2017","journal-title":"Clin Genet"},{"issue":"2","key":"pone.0284409.ref016","doi-asserted-by":"crossref","first-page":"243","DOI":"10.1038\/jhg.2016.116","article-title":"The sensitivity of exome sequencing in identifying pathogenic mutations for LGMD in the United States","volume":"62","author":"HM Reddy","year":"2017","journal-title":"J Hum Genet"},{"issue":"9","key":"pone.0284409.ref017","doi-asserted-by":"crossref","first-page":"1478","DOI":"10.1038\/s41436-020-0840-3","article-title":"Sequential targeted exome sequencing of 1001 patients affected by unexplained limb-girdle weakness","volume":"22","author":"A T\u00f6pf","year":"2020","journal-title":"Genet Med"},{"key":"pone.0284409.ref018","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.cca.2017.07.002","article-title":"Prevalence of anti-NT5C1A antibodies in Japanese patients with autoimmune rheumatic diseases in comparison with other patient cohorts","volume":"472","author":"Y Muro","year":"2017","journal-title":"Clin Chim Acta"},{"issue":"5","key":"pone.0284409.ref019","doi-asserted-by":"crossref","DOI":"10.3390\/cells10051146","article-title":"Anti-cN1A Antibodies Are Associated with More Severe Dysphagia in Sporadic Inclusion Body Myositis","volume":"10","author":"M Lucchini","year":"2021","journal-title":"Cells"},{"issue":"4","key":"pone.0284409.ref020","doi-asserted-by":"crossref","first-page":"496","DOI":"10.1002\/mus.26813","article-title":"Quantitative muscle MRI study of patients with sporadic inclusion body myositis","volume":"61","author":"B Ansari","year":"2020","journal-title":"Muscle Nerve"},{"issue":"6","key":"pone.0284409.ref021","doi-asserted-by":"crossref","first-page":"1153","DOI":"10.1093\/rheumatology\/ker001","article-title":"Magnetic resonance imaging of skeletal muscles in sporadic inclusion body myositis","volume":"50","author":"FM Cox","year":"2011","journal-title":"Rheumatology (Oxford)"},{"issue":"6","key":"pone.0284409.ref022","doi-asserted-by":"crossref","first-page":"956","DOI":"10.1002\/mus.24661","article-title":"Magnetic resonance imaging pattern recognition in sporadic inclusion-body myositis","volume":"52","author":"G Tasca","year":"2015","journal-title":"Muscle Nerve"},{"issue":"3","key":"pone.0284409.ref023","doi-asserted-by":"crossref","first-page":"e12954","DOI":"10.1111\/bpa.12954","article-title":"MRI and muscle imaging for idiopathic inflammatory myopathies","volume":"31","author":"S Malartre","year":"2021","journal-title":"Brain Pathol"},{"issue":"1","key":"pone.0284409.ref024","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1016\/j.autrev.2009.03.005","article-title":"Use of a commercial line blot assay as a screening test for autoantibodies in inflammatory myopathies","volume":"9","author":"J R\u00f6nnelid","year":"2009","journal-title":"Autoimmun Rev"},{"issue":"3","key":"pone.0284409.ref025","doi-asserted-by":"crossref","first-page":"150","DOI":"10.1016\/j.autrev.2010.09.005","article-title":"Clinical and laboratory aspects of Ro\/SSA-52 autoantibodies","volume":"10","author":"C Defendenti","year":"2011","journal-title":"Autoimmun Rev"},{"issue":"4","key":"pone.0284409.ref026","doi-asserted-by":"crossref","first-page":"289","DOI":"10.1016\/j.nmd.2014.12.009","article-title":"Targeted sequencing and identification of genetic variants in sporadic inclusion body myositis","volume":"25","author":"CC Weihl","year":"2015","journal-title":"Neuromuscul Disord"},{"key":"pone.0284409.ref027","doi-asserted-by":"crossref","first-page":"218.e1","DOI":"10.1016\/j.neurobiolaging.2016.07.024","article-title":"Rare variants in SQSTM1 and VCP genes and risk of sporadic inclusion body myositis","volume":"47","author":"Q Gang","year":"2016","journal-title":"Neurobiol Aging"}],"container-title":["PLOS ONE"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0284409","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,4,14]],"date-time":"2023-04-14T17:31:48Z","timestamp":1681493508000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0284409"}},"subtitle":[],"editor":[{"given":"Latika","family":"Gupta","sequence":"first","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2023,4,14]]},"references-count":27,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2023,4,14]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pone.0284409","relation":{},"ISSN":["1932-6203"],"issn-type":[{"value":"1932-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,4,14]]}}}