{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T15:19:23Z","timestamp":1786979963263,"version":"build-2736575974"},"reference-count":26,"publisher":"Springer Science and Business Media LLC","issue":"6","license":[{"start":{"date-parts":[[2021,4,8]],"date-time":"2021-04-08T00:00:00Z","timestamp":1617840000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,4,8]],"date-time":"2021-04-08T00:00:00Z","timestamp":1617840000000},"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":["Rheumatol Int"],"published-print":{"date-parts":[[2021,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Immunoglobulin G4-related disease (IgG4-RD) is a recently recognized fibro-inflammatory pathology that has been reported to affect principally the retroperitoneum, hepatobiliary system, salivary glands, orbital structures or lymph nodes. However, IgG4-RD with laryngeal involvement is a very rare entity. Our aims were to describe a case of subglottic stenosis as first and only manifestation of IgG4-RD and review the literature. A patient with IgG4-RD affecting the larynx that presented as subglottic stenosis is described. A MEDLINE database search of IgG4-RD cases with laryngopharyngeal manifestations was also conducted. A 30-year-old Caucasian woman was referred to a tertiary care hospital for dyspnea on exertion, which had been increasing for the last 4\u00a0months. Medical and surgical procedures revealed a subglottic stenosis, with a histological finding of IgG4 positive plasma cell infiltration. There was no evidence of other organ involvement. She was successfully treated with oral glucocorticoids and rituximab infusions. Glucocorticoids were rapidly tapered and the rituximab regimen was optimized, with no evidence of relapses. In the literature review, we found a total of 12 reported cases with laryngopharyngeal involvement, two of them with subglottic stenosis. IgG4-RD of the larynx is rare but should be considered after excluding more common disorders.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1007\/s00296-021-04816-4","type":"journal-article","created":{"date-parts":[[2021,4,8]],"date-time":"2021-04-08T15:07:39Z","timestamp":1617894459000},"page":"1161-1167","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Atypical presentation of immunoglobulin G4-related disease as subglottic stenosis: a case-based review"],"prefix":"10.1007","volume":"41","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7119-0786","authenticated-orcid":false,"given":"Bel\u00e9n","family":"Atienza-Mateo","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teresa","family":"D\u00edaz de Ter\u00e1n-L\u00f3pez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2849-9435","authenticated-orcid":false,"given":"Javier","family":"G\u00f3mez-Rom\u00e1n","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"S\u00e1nchez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roberto","family":"Mons-Lera","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio","family":"Rubio-Su\u00e1rez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3682-0142","authenticated-orcid":false,"given":"Jos\u00e9 Manuel","family":"Cifri\u00e1n","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7924-7406","authenticated-orcid":false,"given":"Miguel \u00c1.","family":"Gonz\u00e1lez-Gay","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,4,8]]},"reference":[{"issue":"9","key":"4816_CR1","doi-asserted-by":"publisher","first-page":"591","DOI":"10.1016\/j.autrev.2010.05.003","volume":"9","author":"H Takahashi","year":"2010","unstructured":"Takahashi H, Yamamoto M, Suzuki C, Naishiro Y, Shinomura Y, Imai K (2010) The birthday of a new syndrome: IgG4-related diseases constitute a clinical entity. Autoimmun Rev 9(9):591\u2013594. https:\/\/doi.org\/10.1016\/j.autrev.2010.05.003","journal-title":"Autoimmun Rev"},{"issue":"10","key":"4816_CR2","doi-asserted-by":"publisher","first-page":"3061","DOI":"10.1002\/art.34593","volume":"64","author":"JH Stone","year":"2012","unstructured":"Stone JH, Khosroshahi A, Deshpande V, Chan JK, Heathcote JG, Aalberse R et al (2012) Recommendations for the nomenclature of IgG4-related disease and its individual organ system manifestations. Arthritis Rheum 64(10):3061\u20133067. https:\/\/doi.org\/10.1002\/art.34593","journal-title":"Arthritis Rheum"},{"issue":"9","key":"4816_CR3","doi-asserted-by":"publisher","first-page":"1181","DOI":"10.1038\/modpathol.2012.72","volume":"25","author":"V Deshpande","year":"2012","unstructured":"Deshpande V, Zen Y, Chan JK, Yi EE, Sato Y, Yoshino T et al (2012) Consensus statement on the pathology of IgG4-related disease. Mod Pathol 25(9):1181\u20131192. https:\/\/doi.org\/10.1038\/modpathol.2012.72","journal-title":"Mod Pathol"},{"key":"4816_CR4","unstructured":"Nambiar S, Oliver TI (2020) IgG4 Related Disease. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan."},{"issue":"1","key":"4816_CR5","doi-asserted-by":"publisher","first-page":"108","DOI":"10.1097\/BOR.0b013e3283413469","volume":"23","author":"RP Sah","year":"2011","unstructured":"Sah RP, Chari ST (2011) Serologic issues in IgG4-related systemic disease and autoimmune pancreatitis. Curr Opin Rheumatol 23(1):108\u2013113. https:\/\/doi.org\/10.1097\/BOR.0b013e3283413469","journal-title":"Curr Opin Rheumatol"},{"issue":"9","key":"4816_CR6","doi-asserted-by":"publisher","first-page":"2466","DOI":"10.1002\/art.39205","volume":"67","author":"ZS Wallace","year":"2015","unstructured":"Wallace ZS, Deshpande V, Mattoo H, Mahajan VS, Kulikova M, Pillai S et al (2015) IgG4-related disease: clinical and laboratory features in one hundred twenty-five patients. Arthritis Rheumatol 67(9):2466\u20132475. https:\/\/doi.org\/10.1002\/art.39205","journal-title":"Arthritis Rheumatol"},{"issue":"1","key":"4816_CR7","doi-asserted-by":"publisher","first-page":"83","DOI":"10.1097\/BOR.0000000000000133","volume":"27","author":"ZS Wallace","year":"2015","unstructured":"Wallace ZS, Stone JH (2015) An update on IgG4-related disease. Curr Opin Rheumatol 27(1):83\u201390. https:\/\/doi.org\/10.1097\/BOR.0000000000000133","journal-title":"Curr Opin Rheumatol"},{"issue":"9","key":"4816_CR8","doi-asserted-by":"publisher","first-page":"597","DOI":"10.1016\/j.arbres.2020.04.013","volume":"56","author":"M Tello-S\u00e1nchez","year":"2020","unstructured":"Tello-S\u00e1nchez M, Rodr\u00edguez-Duque MS, Loidi-L\u00f3pez C, Mart\u00edn-Arroyo J, Gonz\u00e1lez-Gay M\u00c1, Fern\u00e1ndez-Ayala Novo M (2020) Pleural and pericardial effusion as the only manifestation of IgG4-related disease. Arch Bronconeumol 56(9):597\u2013599. https:\/\/doi.org\/10.1016\/j.arbres.2020.04.013","journal-title":"Arch Bronconeumol"},{"issue":"6","key":"4816_CR9","doi-asserted-by":"publisher","first-page":"1538","DOI":"10.1002\/hep.21697","volume":"45","author":"Y Zen","year":"2007","unstructured":"Zen Y, Fujii T, Harada K, Kawano M, Yamada K, Takahira M et al (2007) Th2 and regulatory immune reactions are increased in immunoglobin G4-related sclerosing pancreatitis and cholangitis. Hepatology 45(6):1538\u20131546. https:\/\/doi.org\/10.1002\/hep.21697","journal-title":"Hepatology"},{"issue":"4","key":"4816_CR10","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1111\/j.1365-2222.2009.03207.x","volume":"39","author":"RC Aalberse","year":"2009","unstructured":"Aalberse RC, Stapel SO, Schuurman J, Rispens T (2009) Immunoglobulin G4: an odd antibody. Clin Exp Allergy 39(4):469\u2013477. https:\/\/doi.org\/10.1111\/j.1365-2222.2009.03207.x","journal-title":"Clin Exp Allergy"},{"issue":"4","key":"4816_CR11","first-page":"203","volume":"10","author":"R Opri\u0163\u0103","year":"2017","unstructured":"Opri\u0163\u0103 R, Opri\u0163\u0103 B, Berceanu D, Diaconescu IB (2017) Overview of IgG4 - related disease. J Med Life 10(4):203\u2013207","journal-title":"J Med Life"},{"issue":"7","key":"4816_CR12","doi-asserted-by":"publisher","first-page":"1688","DOI":"10.1002\/art.39132","volume":"67","author":"A Khosroshahi","year":"2015","unstructured":"Khosroshahi A, Wallace ZS, Crowe JL, Akamizu T, Azumi A, Carruthers MN et al (2015) International consensus guidance statement on the management and treatment of IgG4-related disease. Arthritis Rheumatol 67(7):1688\u20131699. https:\/\/doi.org\/10.1002\/art.39132","journal-title":"Arthritis Rheumatol"},{"issue":"6","key":"4816_CR13","doi-asserted-by":"publisher","first-page":"1171","DOI":"10.1136\/annrheumdis-2014-206605","volume":"74","author":"MN Carruthers","year":"2015","unstructured":"Carruthers MN, Topazian MD, Khosroshahi A, Witzig TE, Wallace ZS, Hart PA et al (2015) Rituximab for IgG4-related disease: a prospective, open-label trial. Ann Rheum Dis 74(6):1171\u20131177. https:\/\/doi.org\/10.1136\/annrheumdis-2014-206605","journal-title":"Ann Rheum Dis"},{"issue":"7","key":"4816_CR14","doi-asserted-by":"publisher","first-page":"681","DOI":"10.1007\/s00393-016-0142-y","volume":"75","author":"CA Perugino","year":"2016","unstructured":"Perugino CA, Stone JH (2016) Treatment of IgG4-related disease: Current and future approaches. Z Rheumatol 75(7):681\u2013686. https:\/\/doi.org\/10.1007\/s00393-016-0142-y","journal-title":"Z Rheumatol"},{"issue":"1","key":"4816_CR15","doi-asserted-by":"publisher","first-page":"77","DOI":"10.1136\/annrheumdis-2019-216561","volume":"79","author":"ZS Wallace","year":"2020","unstructured":"Wallace ZS, Naden RP, Chari S, Choi HK, Della-Torre E, Dicaire JF et al (2020) The 2019 American College of Rheumatology\/European League Against Rheumatism classification criteria for IgG4-related disease. Ann Rheum Dis 79(1):77\u201387. https:\/\/doi.org\/10.1136\/annrheumdis-2019-216561","journal-title":"Ann Rheum Dis"},{"issue":"10","key":"4816_CR16","doi-asserted-by":"publisher","first-page":"1106","DOI":"10.1017\/S0022215110001143","volume":"124","author":"L Masterson","year":"2010","unstructured":"Masterson L, Del Pero MM, Donnelly N, Moffat DA, Rytina E (2010) Immunoglobulin G4 related systemic sclerosing disease involving the temporal bone. J Laryngol Otol 124(10):1106\u20131110. https:\/\/doi.org\/10.1017\/S0022215110001143","journal-title":"J Laryngol Otol"},{"issue":"11","key":"4816_CR17","doi-asserted-by":"publisher","first-page":"1573","DOI":"10.1002\/hed.21232","volume":"32","author":"HU V\u00f6lker","year":"2010","unstructured":"V\u00f6lker HU, Scheich M, Zettl A, Hagen R, M\u00fcller-Hermelink HK, Gattenl\u00f6hner S (2010) Laryngeal inflammatory myofibroblastic tumors: Different clinical appearance and histomorphologic presentation of one entity. Head Neck 32(11):1573\u20131578. https:\/\/doi.org\/10.1002\/hed.21232","journal-title":"Head Neck"},{"issue":"9","key":"4816_CR18","doi-asserted-by":"publisher","first-page":"864","DOI":"10.1001\/archoto.2012.1821","volume":"138","author":"JS Virk","year":"2012","unstructured":"Virk JS, Stamatoglou C, Kwame I, Salama A, Sandison A, Sandhu G (2012) IgG4-sclerosing pseudotumor of the trachea: a case report and review of the literature. Arch Otolaryngol Head Neck Surg 138(9):864\u2013866. https:\/\/doi.org\/10.1001\/archoto.2012.1821","journal-title":"Arch Otolaryngol Head Neck Surg"},{"key":"4816_CR19","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2013-009158","author":"Y Shaib","year":"2013","unstructured":"Shaib Y, Ton E, Goldschmeding R, Tekstra J (2013) IgG4-related disease with atypical laryngeal presentation and Beh\u00e7et\/granulomatous polyangiitis mimicking features. BMJ Case Rep. https:\/\/doi.org\/10.1136\/bcr-2013-009158","journal-title":"BMJ Case Rep"},{"issue":"6","key":"4816_CR20","doi-asserted-by":"publisher","first-page":"E57","DOI":"10.1002\/hed.23519","volume":"36","author":"JF Khoo","year":"2014","unstructured":"Khoo JF, Batt M, Stimpson P, Safdar A (2014) Supraglottic immunoglobulin-G4 related plasma cell granuloma: case report and literature review. Head Neck 36(6):E57\u2013E59. https:\/\/doi.org\/10.1002\/hed.23519","journal-title":"Head Neck"},{"issue":"3","key":"4816_CR21","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1177\/0003489414549574","volume":"124","author":"L Reder","year":"2015","unstructured":"Reder L, Della-Torre E, Stone JH, Mori M, Song P (2015) Clinical Manifestations of IgG4-Related Disease in the Pharynx: Case Series and Review of the Literature. Ann Otol Rhinol Laryngol 124(3):173\u2013178. https:\/\/doi.org\/10.1177\/0003489414549574","journal-title":"Ann Otol Rhinol Laryngol"},{"issue":"2","key":"4816_CR22","doi-asserted-by":"publisher","first-page":"77","DOI":"10.17116\/otorino201782277-79","volume":"82","author":"DM Mustafaev","year":"2017","unstructured":"Mustafaev DM (2017) IgG4-related sclerosing disease of the larynx. Vestn Otorinolaringol. 82(2):77\u201379. https:\/\/doi.org\/10.17116\/otorino201782277-79","journal-title":"Vestn Otorinolaringol."},{"issue":"5 Pt 2","key":"4816_CR23","doi-asserted-by":"publisher","first-page":"416","DOI":"10.1016\/j.reuma.2018.08.011","volume":"16","author":"S Su\u00e1rez-D\u00edaz","year":"2020","unstructured":"Su\u00e1rez-D\u00edaz S, N\u00fa\u00f1ez-Batalla F, Fern\u00e1ndez-Garc\u00eda MS, Fern\u00e1ndez-Llana MB, Yllera-Guti\u00e9rrez C, Caminal-Montero L (2020) Aphthous stomatitis and laryngitis, another form of presentation of an IgG4-related disease? Reumatol Clin 16(5 Pt 2):416\u2013418. https:\/\/doi.org\/10.1016\/j.reuma.2018.08.011","journal-title":"Reumatol Clin"},{"issue":"2","key":"4816_CR24","doi-asserted-by":"publisher","first-page":"305","DOI":"10.1016\/j.anl.2019.05.008","volume":"47","author":"K Matsushima","year":"2020","unstructured":"Matsushima K, Ohira S, Matsui H, Fukuo A, Honma N, Wada K et al (2020) IgG4-related disease with pseudotumor formation in the larynx. Auris Nasus Larynx 47(2):305\u2013308. https:\/\/doi.org\/10.1016\/j.anl.2019.05.008","journal-title":"Auris Nasus Larynx"},{"issue":"7","key":"4816_CR25","doi-asserted-by":"publisher","first-page":"702","DOI":"10.3760\/cma.j.cn115330-20200106-00007","volume":"55","author":"H Zhong","year":"2020","unstructured":"Zhong H, Li Y, Qiu LL, Wen WP, Lei WB (2020) The invasive IgG4-related disease in larynx: one case report. Chinese J Otorhinolaryngol Head Neck Surgery. 55(7):702\u2013704. https:\/\/doi.org\/10.3760\/cma.j.cn115330-20200106-00007","journal-title":"Chinese J Otorhinolaryngol Head Neck Surgery."},{"key":"4816_CR26","doi-asserted-by":"publisher","unstructured":"Syed AS, Colombo RE, Syed BS, Henning PM (2020) Treatment-resistant cough: a rare manifestation of IgG4-related disease involving the larynx. BMJ Case Rep 23;13(11):e237614. Doi: https:\/\/doi.org\/10.1136\/bcr-2020-237614","DOI":"10.1136\/bcr-2020-237614"}],"container-title":["Rheumatology International"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00296-021-04816-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00296-021-04816-4\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00296-021-04816-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,4,27]],"date-time":"2021-04-27T11:14:56Z","timestamp":1619522096000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00296-021-04816-4"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,4,8]]},"references-count":26,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2021,6]]}},"alternative-id":["4816"],"URL":"https:\/\/doi.org\/10.1007\/s00296-021-04816-4","relation":{},"ISSN":["0172-8172","1437-160X"],"issn-type":[{"value":"0172-8172","type":"print"},{"value":"1437-160X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,4,8]]},"assertion":[{"value":"5 January 2021","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"16 February 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"8 April 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Compliance with ethical standards"}},{"value":"The authors report no conflicts of interest related to this manuscript.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"Informed consent was obtained from the patient described in the study, including permission for publication of images.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed consent"}}]}}