{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,28]],"date-time":"2026-01-28T20:23:30Z","timestamp":1769631810564,"version":"3.49.0"},"reference-count":13,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2020,4,7]],"date-time":"2020-04-07T00:00:00Z","timestamp":1586217600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2020,4,7]],"date-time":"2020-04-07T00:00:00Z","timestamp":1586217600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"This work was supported in part by Takeda Science Foundation."}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Endocr Disord"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n<jats:title>Background<\/jats:title>\n<jats:p>Autoimmune polyglandular syndrome type 2 (APS-2) is a rare and complex clinical entity, and little is known about its etiology and progression.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Case presentation<\/jats:title>\n<jats:p>A 52-year-old woman with autoimmune hepatitis (AIH) and bronchial asthma was diagnosed with APS-2; autoimmune Addison\u2019s disease (AD), and Hashimoto\u2019s thyroiditis (HT), and she underwent prednisolone (PSL) treatment. Five months later, she presented ptosis and was diagnosed with thymoma-associated myasthenia gravis (MG). Thymectomy and PSL treatment with immuno-suppressants appeared to ameliorate MG, AD, AIH, HT, and bronchial asthma. HLA typing analysis revealed that the patient had susceptible HLA alleles to MG, AIH, and HT in a Japanese population.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>This case suggests common endocrinological and autoimmune aspects of APS-2 and AIH with thymoma-associated MG, which are considered to be extremely rare complications.<\/jats:p>\n<\/jats:sec>","DOI":"10.1186\/s12902-020-0498-5","type":"journal-article","created":{"date-parts":[[2020,4,7]],"date-time":"2020-04-07T14:03:04Z","timestamp":1586268184000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Autoimmune polyglandular syndrome type 2 and autoimmune hepatitis with thymoma-associated myasthenia gravis: case report"],"prefix":"10.1186","volume":"20","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7718-734X","authenticated-orcid":false,"given":"Hidefumi","family":"Inaba","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroyuki","family":"Ariyasu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroshi","family":"Iwakura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chiaki","family":"Kurimoto","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoko","family":"Ueda","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shinsuke","family":"Uraki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ken","family":"Takeshima","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasushi","family":"Furukawa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shuhei","family":"Morita","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshiaki","family":"Nakayama","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuya","family":"Ohashi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hidefumi","family":"Ito","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshiharu","family":"Nishimura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takashi","family":"Akamizu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,4,7]]},"reference":[{"issue":"3","key":"498_CR1","doi-asserted-by":"publisher","first-page":"327","DOI":"10.1210\/edrv.23.3.0466","volume":"23","author":"C Betterle","year":"2002","unstructured":"Betterle C, Dal Pra C, Mantero F, Zanchetta R. Autoimmune adrenal insufficiency and autoimmune polyendocrine syndromes: autoantibodies, autoantigens, and their applicability in diagnosis and disease prediction. Endocr RevEndocr Rev. 2002;23(3):327\u201364.","journal-title":"Endocr RevEndocr Rev"},{"key":"498_CR2","doi-asserted-by":"publisher","unstructured":"Romi F. Thymoma in Myasthenia Gravis: From Diagnosis to Treatment. Autoimmune Dis. 2011;474512. https:\/\/doi.org\/10.4061\/2011\/474512.","DOI":"10.4061\/2011\/474512"},{"issue":"3","key":"498_CR3","doi-asserted-by":"publisher","first-page":"121","DOI":"10.3109\/00207454.2010.539307","volume":"121","author":"ZF Mao","year":"2011","unstructured":"Mao ZF, Yang LX, Mo XA, Qin C, Lai YR, He NY, Li T, Hackett ML. Frequency of autoimmune diseases in myasthenia gravis: a systematic review. Int J Neurosci. 2011;121(3):121\u20139.","journal-title":"Int J Neurosci"},{"issue":"4","key":"498_CR4","doi-asserted-by":"publisher","first-page":"578","DOI":"10.1016\/j.jhep.2004.12.019","volume":"42","author":"K Yoshizawa","year":"2005","unstructured":"Yoshizawa K, Ota M, Katsuyama Y, Ichijo T, Matsumoto A, Tanaka E, Kiyosawa K. Genetic analysis of the HLA region of Japanese patients with type 1 autoimmune hepatitis. J HepatolJ Hepatol. 2005;42(4):578\u201384.","journal-title":"J HepatolJ Hepatol"},{"key":"498_CR5","first-page":"292","volume":"60","author":"H Asakawa","year":"2002","unstructured":"Asakawa H, Kashihara T, Fukuda H, Yamamoto M. A patient with thymoma and four different organ-specific autoimmune diseases. Neth J MedNeth J Med. 2002;60:292\u20135.","journal-title":"Neth J MedNeth J Med"},{"issue":"6","key":"498_CR6","doi-asserted-by":"publisher","first-page":"361","DOI":"10.1097\/NRL.0b013e3181945437","volume":"15","author":"S Konno","year":"2009","unstructured":"Konno S, Ichijo T, Murata M, Toda T, Nakazora H, Nomoto N, Sugimoto H, Nemoto H, Kurihara T, Wakata N, et al. Autoimmune polyglandular syndrome type 2 with myasthenia gravis crisis. Neurologist. 2009;15(6):361\u20133.","journal-title":"Neurologist"},{"issue":"2","key":"498_CR7","doi-asserted-by":"publisher","first-page":"E379","DOI":"10.1210\/jc.2013-2841","volume":"99","author":"S Ueda","year":"2014","unstructured":"Ueda S, Oryoji D, Yamamoto K, Noh JY, Okamura K, Noda M, Kashiwase K, Kosuga Y, Sekiya K, Inoue K, et al. Identification of independent susceptible and protective HLA alleles in Japanese autoimmune thyroid disease and their epistasis. J Clin Endocrinol MetabJ Clin Endocrinol Metab. 2014;99(2):E379\u201383.","journal-title":"J Clin Endocrinol MetabJ Clin Endocrinol Metab"},{"issue":"1","key":"498_CR8","doi-asserted-by":"publisher","first-page":"49","DOI":"10.3109\/08916939409008008","volume":"19","author":"T Horiki","year":"1994","unstructured":"Horiki T, Inoko H, Moriuchi J, Ichikawa Y, Arimori S. Combinations of HLA-DPB1 and HLA-DQB1 alleles determine susceptibility to early-onset myasthenia gravis in Japan. Autoimmunity. 1994;19(1):49\u201354.","journal-title":"Autoimmunity"},{"issue":"Suppl 2","key":"498_CR9","first-page":"5","volume":"31","author":"HK M\u00fcller-Hermelink","year":"1999","unstructured":"M\u00fcller-Hermelink HK, Marx A. Pathological aspects of malignant and benign thymic disorders. Ann MedAnn Med. 1999;31(Suppl 2):5\u201314.","journal-title":"Ann MedAnn Med"},{"issue":"5","key":"498_CR10","doi-asserted-by":"publisher","first-page":"367","DOI":"10.3816\/CLC.2009.n.051","volume":"10","author":"M Seker","year":"2009","unstructured":"Seker M, Gozu HI, Oven Ustaalio\u011flu BB, Sonmez B, Erkal FY, Kocak M, Barisik NO, Orbay E, Sargin M, Sargin H, et al. Myasthenia gravis and autoimmune Addison disease in a patient with thymoma. Clin Lung Cancer. 2009;10(5):367\u201370.","journal-title":"Clin Lung Cancer"},{"key":"498_CR11","doi-asserted-by":"publisher","first-page":"80","DOI":"10.12659\/MSMBR.900193","volume":"22","author":"Y Yao","year":"2016","unstructured":"Yao Y, Zhu L, Li J, Jin Y, He L. Association of HLA-DRB1 gene polymorphism with risk of asthma: a meta-analysis. Med Sci Monit Basic ResMed Sci Monit Basic Res. 2016;22:80\u20136.","journal-title":"Med Sci Monit Basic ResMed Sci Monit Basic Res"},{"issue":"1","key":"498_CR12","doi-asserted-by":"publisher","first-page":"82","DOI":"10.1016\/j.autrev.2015.09.005","volume":"15","author":"C Bernard","year":"2016","unstructured":"Bernard C, Frih H, Pasquet F, Kerever S, Jamilloux Y, Tronc F, Guibert B, Isaac S, Devouassoux M, Chalabreysse L, et al. Thymoma associated with autoimmune diseases: 85 cases and literature review. Autoimmun RevAutoimmun Rev. 2016;15(1):82\u201392.","journal-title":"Autoimmun RevAutoimmun Rev"},{"key":"498_CR13","volume-title":"Endotext [Internet]","author":"T Akamizu","year":"2000","unstructured":"Akamizu T, Amino N. Hashimoto\u2019s Thyroiditis. In: Feingold KR, Anawalt B, Boyce A, Chrousos G, Dungan K, Grossman A, Hershman JM, Kaltsas G, Koch C, Kopp P, et al., editors. Endotext [Internet]. South Dartmouth (MA): MDText.com, Inc; 2000\u2013.2017."}],"container-title":["BMC Endocrine Disorders"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-020-0498-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s12902-020-0498-5\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-020-0498-5.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,4,6]],"date-time":"2021-04-06T23:13:06Z","timestamp":1617750786000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcendocrdisord.biomedcentral.com\/articles\/10.1186\/s12902-020-0498-5"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,4,7]]},"references-count":13,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["498"],"URL":"https:\/\/doi.org\/10.1186\/s12902-020-0498-5","relation":{},"ISSN":["1472-6823"],"issn-type":[{"value":"1472-6823","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,4,7]]},"assertion":[{"value":"16 May 2019","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"24 January 2020","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"7 April 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Not applicable.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent for publication of the case was obtained from the patient. A copy of the consent form is available for review by the editor of the journal.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"All authors (HIN, HA, HIK, CK, YU, SU, KT, YF, SM, YNY, TO, HIT, YNM, and TA) have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"47"}}