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Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review

Immunoglobulin G4 (IgG4)-related disease is characterized by the typical histopathological features of a dense lymphoplasmacytic infiltrate rich in IgG4-positive plasma cells, a high ratio of IgG4- to IgG-positive cells, storiform fibrosis (cellular fibrosis organized in an irregular whorled pattern...

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Autores principales: Oh, Ji Hyun, Lee, Tae Hee, Kim, Hyo Shik, Jung, Chan Sung, Lee, Joon Seong, Hong, Su Jin, Jin, So-Young
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5058890/
https://www.ncbi.nlm.nih.gov/pubmed/26683918
http://dx.doi.org/10.1097/MD.0000000000002122
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author Oh, Ji Hyun
Lee, Tae Hee
Kim, Hyo Shik
Jung, Chan Sung
Lee, Joon Seong
Hong, Su Jin
Jin, So-Young
author_facet Oh, Ji Hyun
Lee, Tae Hee
Kim, Hyo Shik
Jung, Chan Sung
Lee, Joon Seong
Hong, Su Jin
Jin, So-Young
author_sort Oh, Ji Hyun
collection PubMed
description Immunoglobulin G4 (IgG4)-related disease is characterized by the typical histopathological features of a dense lymphoplasmacytic infiltrate rich in IgG4-positive plasma cells, a high ratio of IgG4- to IgG-positive cells, storiform fibrosis (cellular fibrosis organized in an irregular whorled pattern), obliterative phlebitis, and variable presence of eosinophils. The disease exhibits systemic involvement but very rarely involves the esophagus. A 33-year-old man was admitted to our hospital for evaluation of a 1-year history of progressive dysphagia. Neck imaging revealed a 3.9-cm mass in the cervical esophagus and multifocal calcified lymph nodes in the lower neck and mediastinum. Two previous tertiary hospitals failed to diagnose the patient's condition despite the use of ultrasound-guided needle biopsy of the neck tumor. We performed neck imaging studies, a flexible endoscopic swallowing study, high-resolution manometry, upper endoscopy, and a review of the previous pathologic slides. The patient was finally diagnosed with IgG4-related esophagitis and showed a good response to corticosteroid therapy. We herein report a rare case of dysphagia associated with IgG4-related disease and present a review of the literature.
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spelling pubmed-50588902016-11-01 Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review Oh, Ji Hyun Lee, Tae Hee Kim, Hyo Shik Jung, Chan Sung Lee, Joon Seong Hong, Su Jin Jin, So-Young Medicine (Baltimore) 4500 Immunoglobulin G4 (IgG4)-related disease is characterized by the typical histopathological features of a dense lymphoplasmacytic infiltrate rich in IgG4-positive plasma cells, a high ratio of IgG4- to IgG-positive cells, storiform fibrosis (cellular fibrosis organized in an irregular whorled pattern), obliterative phlebitis, and variable presence of eosinophils. The disease exhibits systemic involvement but very rarely involves the esophagus. A 33-year-old man was admitted to our hospital for evaluation of a 1-year history of progressive dysphagia. Neck imaging revealed a 3.9-cm mass in the cervical esophagus and multifocal calcified lymph nodes in the lower neck and mediastinum. Two previous tertiary hospitals failed to diagnose the patient's condition despite the use of ultrasound-guided needle biopsy of the neck tumor. We performed neck imaging studies, a flexible endoscopic swallowing study, high-resolution manometry, upper endoscopy, and a review of the previous pathologic slides. The patient was finally diagnosed with IgG4-related esophagitis and showed a good response to corticosteroid therapy. We herein report a rare case of dysphagia associated with IgG4-related disease and present a review of the literature. Wolters Kluwer Health 2015-12-18 /pmc/articles/PMC5058890/ /pubmed/26683918 http://dx.doi.org/10.1097/MD.0000000000002122 Text en Copyright © 2015 Wolters Kluwer Health, Inc. All rights reserved. http://creativecommons.org/licenses/by/4.0 This is an open access article distributed under the Creative Commons Attribution License 4.0, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0
spellingShingle 4500
Oh, Ji Hyun
Lee, Tae Hee
Kim, Hyo Shik
Jung, Chan Sung
Lee, Joon Seong
Hong, Su Jin
Jin, So-Young
Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review
title Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review
title_full Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review
title_fullStr Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review
title_full_unstemmed Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review
title_short Esophageal Involvement of Immunoglobulin G4-Related Disease: A Case Report and Literature Review
title_sort esophageal involvement of immunoglobulin g4-related disease: a case report and literature review
topic 4500
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5058890/
https://www.ncbi.nlm.nih.gov/pubmed/26683918
http://dx.doi.org/10.1097/MD.0000000000002122
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