{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T10:40:01Z","timestamp":1761302401076,"version":"build-2065373602"},"reference-count":10,"publisher":"Springer Science and Business Media LLC","issue":"11","license":[{"start":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T00:00:00Z","timestamp":1756684800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/deed.de"},{"start":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T00:00:00Z","timestamp":1756684800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/deed.de"}],"funder":[{"DOI":"10.13039\/501100006447","name":"University of Zurich","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100006447","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["HNO"],"published-print":{"date-parts":[[2025,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Eosinophilic granulomatosis with polyangiitis (EGPA, previously known as Churg\u2013Strauss syndrome) is an antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) often presenting with chronic rhinosinusitis, pulmonary infiltrates, gastrointestinal and cardiac disorders, or renal lesions. Sinonasal and inner ear manifestations are common, but other affections of the head and neck area are rarely reported. Here we report a\u00a0case of a\u00a0young woman with a\u00a0histopathological diagnosis of eosinophil-rich granulomatous inflammation affecting solely the tongue without other local or systemic lesions. It suggests histopathologically a\u00a0localized EGPA according to the 1992 Chapel Hill classification but does not formally meet the recent 2022 joint classification criteria for EGPA of the American College of Rheumatology (ACR) and the European Alliance of Associations for Rheumatology (EULAR). In this report, we also describe the difficulty associated with the treatment.<\/jats:p>","DOI":"10.1007\/s00106-025-01662-9","type":"journal-article","created":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T10:02:32Z","timestamp":1756720952000},"page":"799-802","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The woman with a\u00a0swollen tongue","Die Frau mit der geschwollenen Zunge"],"prefix":"10.1007","volume":"73","author":[{"given":"Fabio","family":"Munzinger","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Luca","family":"Giudici","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael B.","family":"Soyka","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tobias","family":"Kleinjung","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daniel","family":"Runggaldier","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2025,9,1]]},"reference":[{"issue":"1","key":"1662_CR1","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1053\/j.ajkd.2019.04.031","volume":"75","author":"D Geetha","year":"2020","unstructured":"Geetha\u00a0D, Jefferson JA (2020) ANCA-Associated Vasculitis: Core Curriculum 2020. Am J Kidney Dis 75(1):124\u2013137. https:\/\/doi.org\/10.1053\/j.ajkd.2019.04.031","journal-title":"Am J Kidney Dis"},{"issue":"3","key":"1662_CR2","doi-asserted-by":"publisher","first-page":"261","DOI":"10.1111\/all.12088","volume":"68","author":"A Vaglio","year":"2013","unstructured":"Vaglio\u00a0A, Buzio C, Zwerina J (2013) Eosinophilic granulomatosis with polyangiitis (Churg-Strauss): state of the art. Allergy 68(3):261\u2013273. https:\/\/doi.org\/10.1111\/all.12088","journal-title":"Allergy"},{"issue":"5","key":"1662_CR3","doi-asserted-by":"publisher","first-page":"603","DOI":"10.1007\/s10157-013-0869-6","volume":"17","author":"JC Jennette","year":"2013","unstructured":"Jennette\u00a0JC (2013) Overview of the 2012 revised International Chapel Hill Consensus Conference nomenclature of vasculitides. Clin Exp Nephrol 17(5):603\u2013606. https:\/\/doi.org\/10.1007\/s10157-013-0869-6","journal-title":"Clin Exp Nephrol."},{"issue":"1","key":"1662_CR4","doi-asserted-by":"publisher","first-page":"11","DOI":"10.3349\/ymj.2022.0435","volume":"64","author":"JY Pyo","year":"2023","unstructured":"Pyo\u00a0JY, Lee LE, Park\u00a0YB, Lee\u00a0SW (2023) Comparison of the 2022 ACR\/EULAR Classification Criteria for Antineutrophil Cytoplasmic Antibody-Associated Vasculitis with Previous Criteria. Yonsei Med\u00a0J 64(1):11\u201317. https:\/\/doi.org\/10.3349\/ymj.2022.0435","journal-title":"Yonsei Med J"},{"issue":"12","key":"1662_CR5","doi-asserted-by":"publisher","first-page":"543","DOI":"10.14740\/jmc2677w","volume":"7","author":"CM Cruz","year":"2016","unstructured":"Cruz\u00a0CM, Oliveira\u00a0J, Sequeira\u00a0T, Ricardo\u00a0M, Magalhaes\u00a0L, Mendonca\u00a0C (2016) The Man With the Swollen Tongue. J\u00a0Med Cases 7(12):543\u2013546","journal-title":"J Med Cases"},{"key":"1662_CR6","doi-asserted-by":"publisher","first-page":"277","DOI":"10.4193\/Rhino14.074","volume":"53","author":"H Petersen","year":"2015","unstructured":"Petersen\u00a0H, G\u00f6tz P, Both M, et al (2015) Manifestation of eosinophilic granulomatosis with polyangiitis in the head and neck. Rhinology 53(3):277\u2013285. https:\/\/doi.org\/10.4193\/Rhino14.074","journal-title":"Rhinology"},{"key":"1662_CR7","doi-asserted-by":"crossref","unstructured":"Noda S, Komiya Y, Soejima M (2022) A case of eosinophilic granulomatosis with polyangiitis presented with blisters on the tongue. Clin Rheumatol 41(6):1925\u20131926","DOI":"10.1007\/s10067-022-06121-2"},{"issue":"1","key":"1662_CR8","doi-asserted-by":"publisher","first-page":"71","DOI":"10.1038\/s41572-020-0204-y","volume":"6","author":"AR Kitching","year":"2020","unstructured":"Kitching\u00a0AR, Anders\u00a0HJ, Basu\u00a0N, et\u00a0al (2020) ANCA-associated vasculitis. Nat Rev Dis Primers 6(1):71. https:\/\/doi.org\/10.1038\/s41572-020-0204-y","journal-title":"Nat Rev Dis Primers"},{"issue":"7","key":"1662_CR9","doi-asserted-by":"publisher","first-page":"617","DOI":"10.1080\/14712598.2019.1623875","volume":"19","author":"D Ennis","year":"2019","unstructured":"Ennis\u00a0D, Lee\u00a0JK, Pagnoux\u00a0C (2019) Mepolizumab for the treatment of eosinophilic granulomatosis with polyangiitis. Expert Opin Biol Ther 19(7):617\u2013630. https:\/\/doi.org\/10.1080\/14712598.2019.1623875","journal-title":"Expert Opin Biol Ther"},{"issue":"1","key":"1662_CR10","doi-asserted-by":"publisher","first-page":"e000905","DOI":"10.1136\/rmdopen-2019-000905","volume":"5","author":"V Teixeira","year":"2019","unstructured":"Teixeira\u00a0V, Mohammad\u00a0AJ, Jones\u00a0RB, Smith\u00a0R, Jayne\u00a0D (2019) Efficacy and safety of rituximab in the treatment of eosinophilic granulomatosis with polyangiitis. RMD Open 5(1):e000905. https:\/\/doi.org\/10.1136\/rmdopen-2019-000905","journal-title":"RMD Open"}],"container-title":["HNO"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00106-025-01662-9.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00106-025-01662-9\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00106-025-01662-9.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T10:34:48Z","timestamp":1761302088000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00106-025-01662-9"}},"subtitle":["A\u00a0rare case of an ANCA-negative localized eosinophil-rich granulomatous inflammation affecting solely the tongue","Ein seltener Fall einer ANCA-negativen lokalisierten eosinophilenreichen granulomat\u00f6sen Entz\u00fcndung der Zunge"],"short-title":[],"issued":{"date-parts":[[2025,9,1]]},"references-count":10,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2025,11]]}},"alternative-id":["1662"],"URL":"https:\/\/doi.org\/10.1007\/s00106-025-01662-9","relation":{},"ISSN":["0017-6192","1433-0458"],"issn-type":[{"type":"print","value":"0017-6192"},{"type":"electronic","value":"1433-0458"}],"subject":[],"published":{"date-parts":[[2025,9,1]]},"assertion":[{"value":"24 June 2025","order":1,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"1 September 2025","order":2,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"F.\u00a0Munzinger, L.\u00a0Giudici, M.B.\u00a0Soyka, T.\u00a0Kleinjung and D.\u00a0Runggaldier declare that they have no competing interests.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"For this article no studies with human participants or animals were performed by any of the authors. All studies mentioned were in accordance with the ethical standards indicated in each case. For images or other information in the manuscript that identifies patients, consent was obtained from them and\/or their legal guardians.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}}]}}