IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review

IgG4-related pleuritis is rare in patients with IgG4-related diseases (IgG4RD). We report a case of IgG4-related pleuritis diagnosed in a 77-year-old Japanese man with right pleural effusion. The pleural effusion exhibited lymphocyte-predominant exudates with elevated adenosine deaminase (ADA) and I...

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Main Authors: Genki Inui, Tomoya Harada, Karen Makishima, Aditya Sri Listyoko, Shunsuke Ohga, Miyu Nishigami, Hiroyuki Tanaka, Hiroki Ishikawa, Hiroki Kohno, Yoshihiro Funaki, Miki Takata, Ryota Okazaki, Masato Morita, Masahiro Kodani, Akira Yamasaki
Format: Article
Language:English
Published: Elsevier 2025-01-01
Series:Respiratory Medicine Case Reports
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Online Access:http://www.sciencedirect.com/science/article/pii/S2213007125000711
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author Genki Inui
Tomoya Harada
Karen Makishima
Aditya Sri Listyoko
Shunsuke Ohga
Miyu Nishigami
Hiroyuki Tanaka
Hiroki Ishikawa
Hiroki Kohno
Yoshihiro Funaki
Miki Takata
Ryota Okazaki
Masato Morita
Masahiro Kodani
Akira Yamasaki
author_facet Genki Inui
Tomoya Harada
Karen Makishima
Aditya Sri Listyoko
Shunsuke Ohga
Miyu Nishigami
Hiroyuki Tanaka
Hiroki Ishikawa
Hiroki Kohno
Yoshihiro Funaki
Miki Takata
Ryota Okazaki
Masato Morita
Masahiro Kodani
Akira Yamasaki
author_sort Genki Inui
collection DOAJ
description IgG4-related pleuritis is rare in patients with IgG4-related diseases (IgG4RD). We report a case of IgG4-related pleuritis diagnosed in a 77-year-old Japanese man with right pleural effusion. The pleural effusion exhibited lymphocyte-predominant exudates with elevated adenosine deaminase (ADA) and IgG4 levels, along with the presence of plasma cells. A pleural biopsy via local anesthesia thoracoscopy (LAT) confirmed the diagnosis of IgG4-related pleuritis, and the patient was treated with prednisolone and azathioprine. Although ADA is a well-known useful marker for diagnosing tuberculous pleuritis, a review of 14 reported cases of IgG4-related pleuritis demonstrated a positive correlation between IgG4 and ADA in pleural effusion (ρ = 0.705, p < 0.05), suggesting that ADA levels could be elevated in IgG4-related pleuritis. Furthermore, all cases with available cytology reports showed the presence of plasma cells, indicating that detecting plasma cells could aid in diagnosis. Pleural biopsy remains the gold standard for the diagnosis of IgG4-related pleuritis. LAT is a safe and effective diagnostic procedure for older patients, enabling direct visualization and biopsy of pleural lesions without intubation. IgG4-related pleuritis presents with specific findings, including dense white granulomatous lesions, vesicular nodular changes, nonspecific inflammatory changes, and pleural thickening. IgG4-positive plasma cells can be detected even in biopsies with diffuse, nonspecific findings. Therefore, LAT is a valuable and safe tool for diagnosing IgG4-related pleuritis. In conclusion, exudative pleural effusion with elevated ADA levels should prompt the consideration of IgG4-related pleuritis in the differential diagnosis. LAT is a minimally invasive and highly accurate diagnostic tool for IgG4-related pleuritis.
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spelling doaj-art-1af3dc1a2b3f47ffba10280e109687fa2025-08-20T02:44:51ZengElsevierRespiratory Medicine Case Reports2213-00712025-01-015610223510.1016/j.rmcr.2025.102235IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature reviewGenki Inui0Tomoya Harada1Karen Makishima2Aditya Sri Listyoko3Shunsuke Ohga4Miyu Nishigami5Hiroyuki Tanaka6Hiroki Ishikawa7Hiroki Kohno8Yoshihiro Funaki9Miki Takata10Ryota Okazaki11Masato Morita12Masahiro Kodani13Akira Yamasaki14Division of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, Japan; Corresponding author.Division of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Pathology, Department of Pathology, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, Japan; Pulmonary and Respiratory Medicine Department, Faculty of Medicine, Brawijaya University-Dr. Saiful Anwar General Hospital, Malang, 65112, IndonesiaDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDepartment of Pulmonology, Kameda Medical Center, Chiba, 296-0041, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, Japan; Cancer Center, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori University, Tottori, 683-8504, JapanIgG4-related pleuritis is rare in patients with IgG4-related diseases (IgG4RD). We report a case of IgG4-related pleuritis diagnosed in a 77-year-old Japanese man with right pleural effusion. The pleural effusion exhibited lymphocyte-predominant exudates with elevated adenosine deaminase (ADA) and IgG4 levels, along with the presence of plasma cells. A pleural biopsy via local anesthesia thoracoscopy (LAT) confirmed the diagnosis of IgG4-related pleuritis, and the patient was treated with prednisolone and azathioprine. Although ADA is a well-known useful marker for diagnosing tuberculous pleuritis, a review of 14 reported cases of IgG4-related pleuritis demonstrated a positive correlation between IgG4 and ADA in pleural effusion (ρ = 0.705, p < 0.05), suggesting that ADA levels could be elevated in IgG4-related pleuritis. Furthermore, all cases with available cytology reports showed the presence of plasma cells, indicating that detecting plasma cells could aid in diagnosis. Pleural biopsy remains the gold standard for the diagnosis of IgG4-related pleuritis. LAT is a safe and effective diagnostic procedure for older patients, enabling direct visualization and biopsy of pleural lesions without intubation. IgG4-related pleuritis presents with specific findings, including dense white granulomatous lesions, vesicular nodular changes, nonspecific inflammatory changes, and pleural thickening. IgG4-positive plasma cells can be detected even in biopsies with diffuse, nonspecific findings. Therefore, LAT is a valuable and safe tool for diagnosing IgG4-related pleuritis. In conclusion, exudative pleural effusion with elevated ADA levels should prompt the consideration of IgG4-related pleuritis in the differential diagnosis. LAT is a minimally invasive and highly accurate diagnostic tool for IgG4-related pleuritis.http://www.sciencedirect.com/science/article/pii/S2213007125000711IgG4-related pleuritisIgG4-related diseaseLocal anesthesia thoracoscopyPleuroscopyMedical thoracoscopyADA
spellingShingle Genki Inui
Tomoya Harada
Karen Makishima
Aditya Sri Listyoko
Shunsuke Ohga
Miyu Nishigami
Hiroyuki Tanaka
Hiroki Ishikawa
Hiroki Kohno
Yoshihiro Funaki
Miki Takata
Ryota Okazaki
Masato Morita
Masahiro Kodani
Akira Yamasaki
IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review
Respiratory Medicine Case Reports
IgG4-related pleuritis
IgG4-related disease
Local anesthesia thoracoscopy
Pleuroscopy
Medical thoracoscopy
ADA
title IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review
title_full IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review
title_fullStr IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review
title_full_unstemmed IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review
title_short IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review
title_sort igg4 related pleuritis diagnosed by local anesthesia thoracoscopy a case report and literature review
topic IgG4-related pleuritis
IgG4-related disease
Local anesthesia thoracoscopy
Pleuroscopy
Medical thoracoscopy
ADA
url http://www.sciencedirect.com/science/article/pii/S2213007125000711
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