{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T03:56:02Z","timestamp":1779335762280,"version":"3.51.4"},"reference-count":15,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2020,7,23]],"date-time":"2020-07-23T00:00:00Z","timestamp":1595462400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2020,7,23]],"date-time":"2020-07-23T00:00:00Z","timestamp":1595462400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["surg case rep"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n<jats:title>Background<\/jats:title>\n<jats:p>IgG4-related disease (IgG4-RD) is a systemic fibro-inflammatory condition that predominantly involves exocrine organs. Concerning its thoracic presentation, it often manifests as interstitial lung disease or fibrosing mediastinitis. It is very rare for IgG4-RD to form a well-defined mass in the anterior mediastinum, mimicking an encapsulated thymoma.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Case presentation<\/jats:title>\n<jats:p>An 82-year-old man with autoimmune pancreatitis under treatment with oral corticosteroids was found to have peripancreatic lymphadenopathy on computed tomography. Subsequent positron emission tomography revealed abnormal uptake (maximal value 3.6) by a thymic mass as well as the peripancreatic lymph nodes. Exacerbation of IgG4-RD was suspected, and we increased the oral steroid dosage. As a result, the peripancreatic lymph nodes, and the anterior mediastinal mass, decreased in size. The thymic mass was suspected to be an encapsulated thymoma because of its lobulated shape, degree of fluorodeoxyglucose accumulation, and response to steroids, and the patient was referred to our department. The serum anti-acetylcholine receptor antibody test was negative. A thoracoscopic tumor resection was performed as diagnostic therapy. Histopathological analysis revealed dense lymphoplasmacytic infiltration with sclerotic stroma within the tumor. Immunohistochemical analysis revealed abundant IgG4-positive plasma cell infiltrates and over 50% IgG4\/IgG-positive plasma cells. We did not see either keratin-positive thymocytes or terminal deoxynucleotidyl transferase-positive lymphocytes. Furthermore, deviation in the kappa chain and lambda chain-positive plasma cells was not noted. Accordingly, IgG4-related sclerosing disease was diagnosed.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>IgG4-related sclerosing masses in the anterior mediastinum are very rare, and the effect of tumor resection on prognosis remains unclear. IgG4-RD had potentially been categorized as Castleman\u2019s disease.<\/jats:p>\n<\/jats:sec>","DOI":"10.1186\/s40792-020-00939-1","type":"journal-article","created":{"date-parts":[[2020,7,23]],"date-time":"2020-07-23T11:04:10Z","timestamp":1595502250000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["A case of IgG4-related anterior mediastinal sclerosing disease coexisting with autoimmune pancreatitis"],"prefix":"10.1186","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2876-6273","authenticated-orcid":false,"given":"Hiroshi","family":"Matsui","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takahiro","family":"Utsumi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Natsumi","family":"Maru","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yohei","family":"Taniguchi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tomohito","family":"Saito","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Haruaki","family":"Hino","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mitsuaki","family":"Ishida","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Koji","family":"Tsuta","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tomohiro","family":"Murakawa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,7,23]]},"reference":[{"key":"939_CR1","doi-asserted-by":"publisher","first-page":"539","DOI":"10.1056\/NEJMra1104650","volume":"366","author":"JH Stone","year":"2012","unstructured":"Stone JH, Zen Y, Deshpande V. IgG4-related disease. N Engl J Med. 2012;366:539\u201351.","journal-title":"N Engl J Med."},{"key":"939_CR2","doi-asserted-by":"publisher","first-page":"117","DOI":"10.1016\/j.rmed.2017.10.005","volume":"132","author":"JP Corcoran","year":"2017","unstructured":"Corcoran JP, Culver EL, Anstey RM, Talwar A, Manganis CD, Cargill TN, et al. Thoracic involvement in IgG4-related disease in a UK-based patient cohort. Respir Med. 2017;132:117\u201321.","journal-title":"Respir Med."},{"key":"939_CR3","doi-asserted-by":"publisher","first-page":"732","DOI":"10.1056\/NEJM200103083441005","volume":"344","author":"H Hamano","year":"2001","unstructured":"Hamano H, Kawa S, Horiuchi A, Unno H, Furuya N, Akamatsu T, et al. High serum IgG4 concentrations in patients with sclerosing pancreatitis. N Engl J Med. 2001;344:732\u20138.","journal-title":"N Engl J Med."},{"key":"939_CR4","doi-asserted-by":"publisher","first-page":"e2150","DOI":"10.1097\/MD.0000000000002150","volume":"94","author":"Y Fei","year":"2015","unstructured":"Fei Y, Shi J, Lin W, Chen Y, Feng R, Wu Q, et al. Intrathoracic involvements of immunoglobulin G4-related sclerosing disease. Medicine (Baltimore). 2015;94:e2150.","journal-title":"Medicine (Baltimore)"},{"key":"939_CR5","doi-asserted-by":"publisher","first-page":"58","DOI":"10.1111\/j.1445-5994.2006.01009.x","volume":"36","author":"K Hirano","year":"2006","unstructured":"Hirano K, Kawabe T, Komatsu Y, Matsubara S, Togawa O, Arizumi T, et al. High-rate pulmonary involvement in autoimmune pancreatitis. Intern Med J. 2006;36:58\u201361.","journal-title":"Intern Med J."},{"key":"939_CR6","doi-asserted-by":"publisher","first-page":"S110","DOI":"10.1097\/JTO.0000000000000295","volume":"9","author":"BW Carter","year":"2014","unstructured":"Carter BW, Okumura M, Detterbeck FC, Marom EM. Approaching the patient with an anterior mediastinal mass: a guide for radiologists. J Thorac Oncol. 2014;9:S110\u20138.","journal-title":"J Thorac Oncol."},{"key":"939_CR7","doi-asserted-by":"publisher","first-page":"888","DOI":"10.1016\/0003-4975(91)90999-7","volume":"51","author":"EW Wilkins Jr","year":"1991","unstructured":"Wilkins EW Jr, Grillo HC, Scannell JG, Moncure AC, Mathisen DJ. J. Maxwell Chamberlain Memorial Paper. Role of staging in prognosis and management of thymoma. Ann Thorac Surg. 1991;51:888\u201392.","journal-title":"Ann Thorac Surg."},{"key":"939_CR8","doi-asserted-by":"publisher","first-page":"381","DOI":"10.1080\/14397595.2017.1290911","volume":"27","author":"H Umehara","year":"2017","unstructured":"Umehara H, Okazaki K, Nakamura T, Satoh-Nakamura T, Nakajima A, Kawano M, et al. Current approach to the diagnosis of IgG4-related disease - Combination of comprehensive diagnostic and organ-specific criteria. Mod Rheumatol. 2017;27:381\u201391.","journal-title":"Mod Rheumatol."},{"key":"939_CR9","doi-asserted-by":"publisher","first-page":"43","DOI":"10.1186\/s13000-016-0493-3","volume":"11","author":"M Nakamura","year":"2016","unstructured":"Nakamura M, Iwamoto O, Chino T, Todoroki K, Kusukawa J. Diagnostic dilemma of IgG4-related primary localized cervical lymphadenopathy associated with aberrant IL-6 expression level. Diagn Pathol. 2016;11:43.","journal-title":"Diagn Pathol."},{"key":"939_CR10","doi-asserted-by":"publisher","first-page":"495","DOI":"10.1097\/PAS.0000000000000575","volume":"40","author":"T Zoshima","year":"2016","unstructured":"Zoshima T, Yamada K, Hara S, Mizushima I, Yamagishi M, Harada K, et al. Multicentric Castleman disease with tubulointerstitial nephritis mimicking IgG4-related disease: Two case reports. Am J Surg Pathol. 2016;40:495\u2013501.","journal-title":"Am J Surg Pathol."},{"key":"939_CR11","doi-asserted-by":"publisher","first-page":"822","DOI":"10.1002\/1097-0142(195607\/08)9:4<822::AID-CNCR2820090430>3.0.CO;2-4","volume":"9","author":"B Castleman","year":"1956","unstructured":"Castleman B, Iverson L, Menendez VP. Localized mediastinal lymphnode hyperplasia resembling thymoma. Cancer. 1956;9:822\u201330.","journal-title":"Cancer."},{"key":"939_CR12","doi-asserted-by":"publisher","first-page":"2627","DOI":"10.1182\/blood-2004-12-4602","volume":"106","author":"N Nishimoto","year":"2005","unstructured":"Nishimoto N, Kanakura Y, Aozasa K, Johkoh T, Nakamura M, Nakano S, et al. Humanized anti-interleukin-6 receptor antibody treatment of multicentric Castleman disease. Blood. 2005;106:2627\u201332.","journal-title":"Blood."},{"key":"939_CR13","doi-asserted-by":"publisher","first-page":"e000432","DOI":"10.1136\/rmdopen-2017-000432","volume":"3","author":"T Sasaki","year":"2017","unstructured":"Sasaki T, Akiyama M, Kaneko Y, Mori T, Yasuoka H, Suzuki K, et al. Distinct features distinguishing IgG4-related disease from multicentric Castleman\u2019s disease. RMD Open. 2017;3:e000432.","journal-title":"RMD Open."},{"key":"939_CR14","doi-asserted-by":"publisher","first-page":"589","DOI":"10.1038\/modpathol.2009.17","volume":"22","author":"Y Sato","year":"2009","unstructured":"Sato Y, Kojima M, Takata K, Morito T, Asaoku H, Takeuchi T, et al. Systemic IgG4-related lymphadenopathy: A clinical and pathologic comparison to multicentric Castleman's disease. Mod Pathol. 2009;22:589\u201399.","journal-title":"Mod Pathol."},{"key":"939_CR15","doi-asserted-by":"publisher","first-page":"1504","DOI":"10.1136\/gut.2008.172908","volume":"58","author":"T Kamisawa","year":"2009","unstructured":"Kamisawa T, Shimosegawa T, Okazaki K, Nishino T, Watanabe H, Kanno A, et al. Standard steroid treatment for autoimmune pancreatitis. Gut. 2009;58:1504\u20137.","journal-title":"Gut."}],"container-title":["Surgical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s40792-020-00939-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s40792-020-00939-1\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s40792-020-00939-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,7,22]],"date-time":"2021-07-22T23:15:03Z","timestamp":1626995703000},"score":1,"resource":{"primary":{"URL":"https:\/\/surgicalcasereports.springeropen.com\/articles\/10.1186\/s40792-020-00939-1"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,7,23]]},"references-count":15,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["939"],"URL":"https:\/\/doi.org\/10.1186\/s40792-020-00939-1","relation":{},"ISSN":["2198-7793"],"issn-type":[{"value":"2198-7793","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,7,23]]},"assertion":[{"value":"20 May 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"14 July 2020","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 July 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Informed consent was obtained for this case report.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and any accompanying images.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no conflicts of interest.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"180"}}