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Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review

We encountered a 78-year-old Japanese man with IgG4-related sialoadenitis complicated with marked eosinophilia. We diagnosed him with IgG4-RD (related disease) with a submandibular gland tumor, serum IgG4 elevation, IgG4-positive plasma cell infiltration, and storiform fibrosis. During follow-up aft...

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Detalles Bibliográficos
Autores principales: Origuchi, Tomoki, Uchida, Tomohisa, Sakaguchi, Tatsuki, Matsuo, Haruna, Michitsuji, Toru, Umeda, Masataka, Shimizu, Toshimasa, Koga, Tomohiro, Kawashiri, Shin-Ya, Iwamoto, Naoki, Ichinose, Kunihiro, Tamai, Mami, Ichinose, Masahiro, Ando, Koji, Horie, Ichiro, Nakao, Nobuhiro, Irie, Junji, Kawakami, Atsushi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Japanese Society of Internal Medicine 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10332968/
https://www.ncbi.nlm.nih.gov/pubmed/36351576
http://dx.doi.org/10.2169/internalmedicine.0453-22
Descripción
Sumario:We encountered a 78-year-old Japanese man with IgG4-related sialoadenitis complicated with marked eosinophilia. We diagnosed him with IgG4-RD (related disease) with a submandibular gland tumor, serum IgG4 elevation, IgG4-positive plasma cell infiltration, and storiform fibrosis. During follow-up after total incision of the submandibular gland, the peripheral eosinophil count was markedly elevated to 29,480 /μL. The differential diagnosis of severe eosinophilia without IgG4-RD was excluded. The patient exhibited a prompt response to corticosteroid therapy. His peripheral blood eosinophil count was the highest ever reported among similar cases. We also review previous cases of IgG4-RD with severe eosinophilia.