{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T04:51:10Z","timestamp":1790743870840,"version":"4.1.0"},"reference-count":26,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2019,3,11]],"date-time":"2019-03-11T00:00:00Z","timestamp":1552262400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100004440","name":"Wellcome Trust","doi-asserted-by":"publisher","award":["085860"],"award-info":[{"award-number":["085860"]}],"id":[{"id":"10.13039\/100004440","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000317","name":"Action Medical Research","doi-asserted-by":"publisher","award":["SP4252"],"award-info":[{"award-number":["SP4252"]}],"id":[{"id":"10.13039\/501100000317","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000341","name":"Arthritis Research UK","doi-asserted-by":"publisher","award":["14518"],"award-info":[{"award-number":["14518"]}],"id":[{"id":"10.13039\/501100000341","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000341","name":"Arthritis Research UK","doi-asserted-by":"publisher","award":["20164"],"award-info":[{"award-number":["20164"]}],"id":[{"id":"10.13039\/501100000341","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000852","name":"Henry Smith Charity","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000852","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000272","name":"National Institute for Health Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000272","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000272","name":"National Institute for Health Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000272","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000765","name":"University College London","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000765","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Neuropathology Appl Neurobio"],"published-print":{"date-parts":[[2019,8]]},"abstract":"<jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>Juvenile idiopathic inflammatory myopathies have been recently reclassified into clinico\u2010serological subgroups. Myopathological correlates of the subgroups are incompletely understood.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We studied muscle biopsies from 101 children with clinically and serologically defined juvenile idiopathic inflammatory myopathies from the UK JDM Cohort and Biomarker Study by applying the international JDM score tool, myopathological review and C5b\u20109 complement analysis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Autoantibody data were available for 90\/101 cases with 18\/90 cases positive for anti\u2010TIF1\u03b3, 15\/90 anti\u2010NXP2, 11\/90 anti\u2010MDA5, 5\/90 anti\u2010Mi2 and 6\/90 anti\u2010PmScl. JDM biopsy severity scores were consistently low in the anti\u2010MDA5 group, high in the anti\u2010Mi2 group, and widely distributed in the other groups. Biopsies were classified histologically as perifascicular atrophy (22\/101), macrophage\u2010rich necrosis (6\/101), scattered necrosis (2\/101), clustered necrosis (2\/101), inflammatory fibre invasion (2\/101), chronic myopathic change (1\/101), diffuse endomysial macrophage infiltrates (40\/101) and minimal change (24\/101). MDA5 cases segregated with the minimal change group and showed no capillary C5b\u20109\u2010deposition. The Mi2 group displayed high severity scores and a tendency towards sarcolemmal complement deposition. NXP2 and TIF1\u03b3 groups showed a variety of pathologies with a high proportion of diffuse endomysial macrophage infiltrates and a high proportion of capillary C5b\u20109 deposition.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>We have shown that juvenile idiopathic inflammatory myopathies have a spectrum of histopathological phenotypes and show distinct complement attack complex deposition patterns. Both correlate in some cases with the serological subtypes. Most cases do not show typical histological features associated with dermatomyositis (e.g. perifascicular atrophy). In contrast, more than half show relatively mild histopathological changes.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/nan.12528","type":"journal-article","created":{"date-parts":[[2018,10,31]],"date-time":"2018-10-31T06:19:25Z","timestamp":1540966765000},"page":"495-512","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":52,"title":["Histological heterogeneity in a large clinical cohort of juvenile idiopathic inflammatory myopathy: analysis by myositis autoantibody and pathological features"],"prefix":"10.1111","volume":"45","author":[{"given":"S. A.","family":"Yasin","sequence":"first","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7962-1560","authenticated-orcid":false,"given":"P. W.","family":"Schutz","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"},{"name":"Division of Neuropathology UCL Institute of Neurology London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C. T.","family":"Deakin","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E.","family":"Sag","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H.","family":"Varsani","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Simou","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L. R.","family":"Marshall","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S. L.","family":"Tansley","sequence":"additional","affiliation":[{"name":"Department of Pharmacy and Pharmacology University of Bath Bath UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N. J.","family":"McHugh","sequence":"additional","affiliation":[{"name":"Department of Pharmacy and Pharmacology University of Bath Bath UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. L.","family":"Holton","sequence":"additional","affiliation":[{"name":"Department of Molecular Neuroscience MRC Centre for Neuromuscular Diseases UCL Institute of Neurology London UK"},{"name":"Division of Neuropathology UCL Institute of Neurology London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L. R.","family":"Wedderburn","sequence":"additional","affiliation":[{"name":"Infection, Immunity, Inflammation Programme UCL GOS Institute of Child Health London UK"},{"name":"Arthritis Research UK Centre for Adolescent Rheumatology at UCL, UCLH and GOSH London UK"},{"name":"NIHR Biomedical Research Centre at Great Ormond Street Hospital London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7833-2158","authenticated-orcid":false,"given":"T. S.","family":"Jacques","sequence":"additional","affiliation":[{"name":"Developmental Biology and Cancer Programme Developmental Biology of Birth Defects UCL GOS Institute of Child Health London UK"},{"name":"Department of Histopathology Great Ormond Street Hospital for Children NHS Foundation Trust London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"the UK Juvenile Dermatomyositis Research Group (UK and Ireland)","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2019,3,11]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/joim.12444"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2004.02.006"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awv192"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.68.4.472"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000001422"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajpath.2015.11.010"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2990.2012.01302.x"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000002863"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/nan.12380"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2017.09.016"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.141443"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.39753"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1186\/ar4600"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12969-017-0205-x"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/acr.23113"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.160871"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1093\/jnen\/62.5.464"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1097\/NEN.0000000000000132"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1136\/annrheumdis-2013-203396"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.23012"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/keq261"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.18637\/jss.v025.i01"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/acr.22620"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM197502202920807"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM197502132920706"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1186\/ar4311"}],"container-title":["Neuropathology and Applied Neurobiology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fnan.12528","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/nan.12528","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,9]],"date-time":"2023-09-09T18:58:49Z","timestamp":1694285929000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/nan.12528"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,3,11]]},"references-count":26,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2019,8]]}},"alternative-id":["10.1111\/nan.12528"],"URL":"https:\/\/doi.org\/10.1111\/nan.12528","archive":["Portico"],"relation":{"has-review":[{"id-type":"doi","id":"10.3410\/f.734414936.793567613","asserted-by":"object"}]},"ISSN":["0305-1846","1365-2990"],"issn-type":[{"value":"0305-1846","type":"print"},{"value":"1365-2990","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,3,11]]},"assertion":[{"value":"2018-01-10","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-10-17","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-03-11","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}