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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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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
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author 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
author_facet 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
author_sort Origuchi, Tomoki
collection PubMed
description 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.
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spelling pubmed-103329682023-07-12 Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review 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 Intern Med Case Report 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. The Japanese Society of Internal Medicine 2022-11-09 2023-06-15 /pmc/articles/PMC10332968/ /pubmed/36351576 http://dx.doi.org/10.2169/internalmedicine.0453-22 Text en Copyright © 2023 by The Japanese Society of Internal Medicine https://creativecommons.org/licenses/by-nc-nd/4.0/The Internal Medicine is an Open Access journal distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit (https://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
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
Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review
title Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review
title_full Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review
title_fullStr Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review
title_full_unstemmed Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review
title_short Immunoglobulin G4-related Disease with Marked Eosinophilia: A Case Report and Literature Review
title_sort immunoglobulin g4-related disease with marked eosinophilia: a case report and literature review
topic Case Report
url 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
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