{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,1]],"date-time":"2025-10-01T15:31:38Z","timestamp":1759332698467,"version":"3.40.5"},"reference-count":16,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2022,11,8]],"date-time":"2022-11-08T00:00:00Z","timestamp":1667865600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["Grant\u2010in\u2010Aid for Scientific Research (16K09674)"],"award-info":[{"award-number":["Grant\u2010in\u2010Aid for Scientific Research (16K09674)"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003478","name":"Ministry of Health, Labour and Welfare","doi-asserted-by":"publisher","award":["Grant\u2010in\u2010Aid for Research on Intractable Diseases"],"award-info":[{"award-number":["Grant\u2010in\u2010Aid for Research on Intractable Diseases"]}],"id":[{"id":"10.13039\/501100003478","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004091","name":"Kumamoto University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004091","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Neuropathology"],"published-print":{"date-parts":[[2023,6]]},"abstract":"<jats:p>Inclusion body myositis (IBM) is a refractory muscle disease characterized by inflammatory and degenerative features in myofibers. Macroglossia is common in systemic amyloid light chain amyloidosis; however, no reports have been published on patients with IBM. We encountered a female patient with clinicopathologically defined IBM who exhibited relatively rapid progression of dysphagia, gait disturbance, and macroglossia. Muscle biopsy demonstrated endomysial mononuclear inflammatory infiltrates, fiber necrosis and regeneration with rimmed vacuoles, and sarcoplasmic inclusions of p62. Tongue biopsy demonstrated fiber degeneration with fatty replacement and fibrosis, nonnecrotic fibers surrounded and invaded by mononuclear cells, and sarcoplasmic dotlike inclusions of p62. Based on the parotid gland, lip, and muscle biopsy, she was diagnosed as having IBM with Sj\u00f6gren's syndrome. She was treated with steroid pulse and intravenous immunoglobulin therapy followed by oral administration of prednisolone, which resulted in temporary clinical improvement. Macroglossia might be an indicator of immunotherapy effectiveness.<\/jats:p>","DOI":"10.1111\/neup.12879","type":"journal-article","created":{"date-parts":[[2022,11,9]],"date-time":"2022-11-09T05:50:49Z","timestamp":1667973049000},"page":"252-256","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Macroglossia in rapidly progressive inclusion body myositis"],"prefix":"10.1111","volume":"43","author":[{"given":"Yoshimune","family":"Yamasaki","sequence":"first","affiliation":[{"name":"Department of Neurology Graduate School of Medical Sciences, Kumamoto University  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7486-0597","authenticated-orcid":false,"given":"Akihiro","family":"Mukaino","sequence":"additional","affiliation":[{"name":"Department of Molecular Neurology and Therapeutics Kumamoto University Hospital  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Satoshi","family":"Yamashita","sequence":"additional","affiliation":[{"name":"Department of Neurology Graduate School of Medical Sciences, Kumamoto University  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yousuke","family":"Takeuchi","sequence":"additional","affiliation":[{"name":"Department of Neurology Graduate School of Medical Sciences, Kumamoto University  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nozomu","family":"Tawara","sequence":"additional","affiliation":[{"name":"Department of Neurology Graduate School of Medical Sciences, Kumamoto University  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryoji","family":"Yoshida","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery, Faculty of Sciences Kumamoto University Hospital  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yumi","family":"Honda","sequence":"additional","affiliation":[{"name":"Department of Diagnostic Pathology Kumamoto University Hospital  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Taro","family":"Yamashita","sequence":"additional","affiliation":[{"name":"Department of Neurology Graduate School of Medical Sciences, Kumamoto University  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Asako","family":"Kakimoto","sequence":"additional","affiliation":[{"name":"Department of Neurology National Hospital Organization Kumamoto Saishun Medical Center  Koshi Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hidetsugu","family":"Ueyama","sequence":"additional","affiliation":[{"name":"Department of Neurology National Hospital Organization Kumamoto Saishun Medical Center  Koshi Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yukio","family":"Ando","sequence":"additional","affiliation":[{"name":"Department of Neurology Graduate School of Medical Sciences, Kumamoto University  Kumamoto Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,11,8]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11926-018-0755-z"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.26830"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clineuro.2011.03.016"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1212\/wnl.48.3.712"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1212\/wnl.58.2.326"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1159\/000330238"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaneurol.2013.3138"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.25058"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.10548"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2012.09.003"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/0003-9993(94)90001-9"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1378\/chest.105.3.947"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1212\/wnl.54.5.1075"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e31826d5ffc"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000002321"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2018-319578"}],"container-title":["Neuropathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/neup.12879","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/neup.12879","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/neup.12879","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,19]],"date-time":"2023-08-19T06:14:50Z","timestamp":1692425690000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/neup.12879"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,11,8]]},"references-count":16,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2023,6]]}},"alternative-id":["10.1111\/neup.12879"],"URL":"https:\/\/doi.org\/10.1111\/neup.12879","archive":["Portico"],"relation":{},"ISSN":["0919-6544","1440-1789"],"issn-type":[{"type":"print","value":"0919-6544"},{"type":"electronic","value":"1440-1789"}],"subject":[],"published":{"date-parts":[[2022,11,8]]},"assertion":[{"value":"2022-06-20","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-10-18","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-11-08","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}