{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,18]],"date-time":"2025-10-18T15:17:56Z","timestamp":1760800676094,"version":"3.37.3"},"reference-count":18,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2023,11,1]],"date-time":"2023-11-01T00:00:00Z","timestamp":1698796800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,12,29]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Eosinophilic granulomatous polyangiitis is a systemic vasculitis associated with bronchial asthma and eosinophilic sinusitis. Here, we describe an unusual presentation of eosinophilic granulomatous polyangiitis that initially manifested as swelling of the oral cavity floor and cervical soft tissue. A 58\u2009year-old Japanese man was diagnosed with bronchial asthma during childhood but did not receive regular medication. Prior to this presentation, he had a persistent cough for over 1\u2009month, and a local physician diagnosed him with bronchial asthma. However, 6\u2009months later, his cough worsened, and a blood test revealed elevated eosinophil levels. Immediately afterward, swelling of the floor of the oral cavity and cervical soft tissue developed. Cellulitis was suspected and antimicrobial treatment was initiated; however, the symptoms persisted and abdominal pain developed. An endoscopic examination revealed duodenitis and a duodenal ulcer. The patient was diagnosed with eosinophilic granulomatous polyangiitis based on three items of the 2022 American College of Rheumatology\/European College of Rheumatology classification criteria: obstructive airway disease, blood eosinophil count \u22651\u2009\u00d7\u2009109 cells\/L, and extravascular eosinophilic infiltration with a score of 10. Oral prednisolone (70\u2009mg\/day), intravenous cyclophosphamide (500\u2009mg\/m2), and subcutaneous mepolizumab (300\u2009mg every 4\u2009weeks) were administered. The patient\u2019s symptoms improved after these treatments, and the eosinophil count and inflammatory marker levels declined. When swelling of the oral cavity floor and cervical soft tissue following an increase in eosinophilia and allergic symptoms occurs, it is crucial to consider the likelihood of eosinophilic granulomatous polyangiitis and collaborate with otolaryngologists and dentists to ensure its prompt identification.<\/jats:p>","DOI":"10.1093\/mrcr\/rxad062","type":"journal-article","created":{"date-parts":[[2023,10,30]],"date-time":"2023-10-30T23:20:30Z","timestamp":1698708030000},"page":"178-181","source":"Crossref","is-referenced-by-count":3,"title":["Eosinophilic granulomatous with polyangiitis complicated by swelling of\u00a0the oral cavity floor and\u00a0cervical soft tissue as initial manifestation mimicking IgG4-related disease: A case report"],"prefix":"10.1093","volume":"8","author":[{"given":"Tomoko","family":"Suzuki","sequence":"first","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mayuko","family":"Moriyama","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ikuko","family":"Takano","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nobue","family":"Miyajima","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuki","family":"Yoshioka","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Manabu","family":"Honda","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2274-7872","authenticated-orcid":false,"given":"Masahiro","family":"Kondo","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sachiko","family":"Shokei","sequence":"additional","affiliation":[{"name":"Pathology Division, Shimane University Hospital , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Asuka","family":"Araki","sequence":"additional","affiliation":[{"name":"Pathology Division, Shimane University Hospital , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kyuichi","family":"Kadota","sequence":"additional","affiliation":[{"name":"Pathology Division, Shimane University Hospital , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8794-7106","authenticated-orcid":false,"given":"Kunihiro","family":"Ichinose","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Shimane University Faculty of Medicine , Shimane, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2023,11,1]]},"reference":[{"key":"2023122911394174900_R1","doi-asserted-by":"crossref","DOI":"10.1016\/j.autrev.2022.103219","article-title":"Eosinophilic granulomatosis with polyangiitis: a review","volume":"22","author":"White","year":"2023","journal-title":"Autoimmun Rev"},{"key":"2023122911394174900_R2","doi-asserted-by":"crossref","first-page":"414","DOI":"10.1002\/1529-0131(200002)43:2<414::AID-ANR23>3.0.CO;2-0","article-title":"Epidemiology of\u00a0systemic vasculitis: a ten-year study in\u00a0the United Kingdom","volume":"43","author":"Watts","year":"2000","journal-title":"Arthritis Rheum"},{"key":"2023122911394174900_R3","doi-asserted-by":"crossref","first-page":"3496","DOI":"10.1002\/art.27678","article-title":"CCL17\/thymus and\u00a0activation-related chemokine in\u00a0Churg-Strauss syndrome","volume":"62","author":"Dallos","year":"2010","journal-title":"Arthritis Rheum"},{"key":"2023122911394174900_R4","doi-asserted-by":"crossref","first-page":"1823","DOI":"10.1093\/rheumatology\/keq445","article-title":"Eotaxin-3 in\u00a0Churg-Strauss syndrome: a clinical and\u00a0immunogenetic study.","volume":"50","author":"Zwerina","year":"2011","journal-title":"Rheumatol (Oxf Engl)"},{"key":"2023122911394174900_R5","doi-asserted-by":"crossref","first-page":"2926","DOI":"10.1002\/art.21250","article-title":"Prevalence and\u00a0clinical significance of\u00a0antineutrophil cytoplasmic antibodies in\u00a0Churg-Strauss syndrome","volume":"52","author":"Sinico","year":"2005","journal-title":"Arthritis Rheum"},{"key":"2023122911394174900_R6","doi-asserted-by":"crossref","first-page":"270","DOI":"10.1002\/art.37721","article-title":"Eosinophilic granulomatosis with polyangiitis (Churg-Strauss): clinical characteristics and\u00a0long-term followup of\u00a0the 383 patients enrolled in\u00a0the French Vasculitis Study Group cohort","volume":"65","author":"Comarmond","year":"2013","journal-title":"Arthritis Rheum"},{"key":"2023122911394174900_R7","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1080\/00016480500437435","article-title":"Ear, nose and\u00a0throat manifestations of\u00a0Churg-Strauss syndrome","volume":"126","author":"Bacciu","year":"2006","journal-title":"Acta Otolaryngol"},{"key":"2023122911394174900_R8","doi-asserted-by":"crossref","first-page":"e377","DOI":"10.1111\/1346-8138.14897","article-title":"Refractory oral ulcers in\u00a0eosinophilic granulomatosis with polyangiitis","volume":"46","author":"Otsuka","year":"2019","journal-title":"J Dermatol"},{"key":"2023122911394174900_R9","doi-asserted-by":"crossref","first-page":"969","DOI":"10.1111\/j.1468-3083.2006.02086.x","article-title":"Oral ulceration: an unusual manifestation of\u00a0Churg-Strauss syndrome","volume":"21","author":"Delplace","year":"2007","journal-title":"J Eur Acad Dermatol Venereol"},{"key":"2023122911394174900_R10","doi-asserted-by":"crossref","first-page":"175","DOI":"10.1016\/j.ajoms.2017.11.007","article-title":"Multiple oral ulcerations: a very rare case of\u00a0Churg-Strauss syndrome with renal disease","volume":"30","author":"Ivanoff","year":"2018","journal-title":"J Oral Maxillofac Surg Med Pathol"},{"key":"2023122911394174900_R11","doi-asserted-by":"crossref","first-page":"1925","DOI":"10.1007\/s10067-022-06121-2","article-title":"A case of\u00a0eosinophilic granulomatosis with polyangiitis presented with blisters on\u00a0the tongue","volume":"41","author":"Noda","year":"2022","journal-title":"Clin Rheumatol"},{"key":"2023122911394174900_R12","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1097\/MD.0b013e318205a4c6","article-title":"The Five-Factor Score revisited: assessment of\u00a0prognoses of\u00a0systemic necrotizing vasculitides based on\u00a0the French Vasculitis Study Group (FVSG) cohort","volume":"90","author":"Guillevin","year":"2011","journal-title":"Med (Baltim)"},{"key":"2023122911394174900_R13","doi-asserted-by":"crossref","first-page":"1359","DOI":"10.1111\/ijd.15986","article-title":"Eosinophilic ulcer of\u00a0the oral mucosa: An update on\u00a0clinicopathologic features, pathogenesis, and\u00a0management","volume":"61","author":"Hamie","year":"2022","journal-title":"Int J Dermatol"},{"key":"2023122911394174900_R14","doi-asserted-by":"crossref","first-page":"416","DOI":"10.1016\/j.cyto.2018.05.009","article-title":"Interleukin-4 contributes to the shift of\u00a0balance of\u00a0IgG subclasses toward IgG4 in\u00a0IgG4-related disease","volume":"110","author":"Akiyama","year":"2018","journal-title":"Cytokine"},{"key":"2023122911394174900_R15","doi-asserted-by":"crossref","DOI":"10.1136\/annrheumdis-2020-218124","article-title":"Eosinophilic granulomatosis with polyangiitis can manifest lacrimal and\u00a0salivary glands swelling by granulomatous inflammation: a potential mimicker of\u00a0IgG4-related disease","volume":"81","author":"Akiyama","year":"2022","journal-title":"Ann Rheum Dis"},{"key":"2023122911394174900_R16","doi-asserted-by":"crossref","DOI":"10.1002\/rcr2.552","article-title":"Eosinophilic granulomatous polyangiitis with IgG4 hypergammaglobulinaemia and\u00a0salivary gland swelling","volume":"8","author":"Takahashi","year":"2020","journal-title":"Respirol Case Rep"},{"key":"2023122911394174900_R17","first-page":"517","article-title":"Churg-Strauss syndrome presenting with conjunctival and\u00a0eyelid masses: a case report","volume":"66","author":"Ameli","year":"2011","journal-title":"Med J Malaysia"},{"key":"2023122911394174900_R18","doi-asserted-by":"crossref","first-page":"2131","DOI":"10.2169\/internalmedicine.52.0674","article-title":"Eosinophilic granulomatosis with polyangiitis of\u00a0the major salivary glands: a case of\u00a0sialadenitis in\u00a0a young patient","volume":"52","author":"Tovoli","year":"2013","journal-title":"Intern Med"}],"container-title":["Modern Rheumatology Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/mrcr\/advance-article-pdf\/doi\/10.1093\/mrcr\/rxad062\/52686351\/rxad062.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/8\/1\/178\/54920080\/rxad062.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/8\/1\/178\/54920080\/rxad062.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,12,29]],"date-time":"2023-12-29T12:28:37Z","timestamp":1703852917000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/mrcr\/article\/8\/1\/178\/7333662"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,11,1]]},"references-count":18,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2023,11,1]]},"published-print":{"date-parts":[[2023,12,29]]}},"URL":"https:\/\/doi.org\/10.1093\/mrcr\/rxad062","relation":{},"ISSN":["2472-5625"],"issn-type":[{"type":"electronic","value":"2472-5625"}],"subject":[],"published-other":{"date-parts":[[2024,1,1]]},"published":{"date-parts":[[2023,11,1]]}}}