Characteristics of pleural effusion in IgG4-related pleuritis.

Published on Jan 1, 2020in Respiratory medicine case reports
· DOI :10.1016/J.RMCR.2020.101019
Zenya Saito5
Estimated H-index: 5
Masahiro Yoshida42
Estimated H-index: 42
+ 2 AuthorsKazuyoshi Kuwano58
Estimated H-index: 58
(Jikei University School of Medicine)
Abstract Here we describe the case of a 78-year-old man with respiratory failure and right pleural effusion. Computed tomography showed right pleural effusion with pleural calcification, tumor-like shadows induced by passive atelectasis, and enlarged mediastinal lymph nodes. Positron emission tomography showed right pleural thickening, rounded atelectasis, and enlarged mediastinal lymph nodes, without fluid accumulation in other organs. The pleural effusion showed lymphocyte-dominated exudates with elevated adenosine deaminase (ADA) levels. Tuberculous pleuritis was suspected, but thoracoscopic pleural biopsy revealed lymphoplasmacytic infiltration and fibrosis, with 10 immunoglobulin G4 (IgG4)-positive plasma cells/high-power field, and IgG4/IgG ratio of 40%. IgG4 concentrations in serum and right pleural effusion were 929 and 1120 mg/dL, respectively. The patient was diagnosed with IgG4-related pleuritis without other systemic manifestations, and reduction in right pleural effusion was confirmed by corticosteroid therapy. IgG4-related disease is typically a systemic disease causing lymphoplasmacytic inflammation in multiple organs. We describe a rare form of IgG4-related pleuritis showing pleural effusion with no other systemic manifestation.
📖 Papers frequently viewed together
14 Citations
5 Citations
10 Citations
#1Toshiyuki KitaH-Index: 15
#2Tomoyuki ArayaH-Index: 9
Last. Kazuo Kasahara (Kanazawa University)H-Index: 26
view all 7 authors...
Abstract A 65-year-old man was admitted for productive cough and dyspnea. Bilateral pleural effusions were observed on chest X-ray. Although the bilateral pleural effusions were exudative with an increased number of lymphocytes, bacterial culture and polymerase chain reaction analysis for Mycobacterium tuberculosis were negative. Immunological examinations showed high levels of immunoglobulin G4 (IgG4) in both serum and pleural effusion fluid. Pathologic evaluation of a left pleural biopsy speci...
11 CitationsSource
#1Atsushi Nagayasu (University of Occupational and Environmental Health Japan)H-Index: 2
#2Satoshi Kubo (University of Occupational and Environmental Health Japan)H-Index: 23
Last. Yoshiya Tanaka (University of Occupational and Environmental Health Japan)H-Index: 93
view all 9 authors...
: An 81-year-old man was admitted with bilateral pleural effusion. A clinical examination showed lymphocytic pleura effusion and elevated serum IgG4 levels, so that IgG4-related disease was suggested, whereas tuberculous pleurisy was suspected because of high adenosine deaminase (ADA) levels in the pleural effusion. A surgical pleural biopsy revealed that there were large numbers of IgG4-positive cells and IgG4/IgG positive cell ratio exceeded 40% in several sites. Accordingly, we diagnosed IgG4...
14 CitationsSource
#1Xiang Tong (Sichuan University)H-Index: 1
#2Min Bai (Sichuan University)H-Index: 2
Last. Hong Fan (Sichuan University)H-Index: 1
view all 6 authors...
Immunoglobulin G4-related disease (IgG4-RD) is a recently described immune-mediated fibroinflammatory disease with a characteristic histopathologic appearance that can affect various organs. We report a 43-year-old Chinese female patient with IgG4-RD involving polyserous effusions with reports of worsening exertional dyspnea for 3 months. Laboratory blood tests revealed that serum interleukin (IL)-6, carbohydrate antigens (CA-199 and CA-125), and alpha-fetoprotein levels were significantly incre...
11 CitationsSource
#1Yoriyuki MurataH-Index: 4
#2Keisuke AoeH-Index: 30
Last. Yusuke MimuraH-Index: 14
view all 10 authors...
: The cause of pleural effusion remains uncertain in approximately 15% of patients despite exhaustive evaluation. As recently described immunoglobulin (Ig)G4-related disease is a fibroinflammatory disorder that can affect various organs, including the lungs, we investigate whether idiopathic pleural effusion includes IgG4-associated etiology. Between 2000 and 2012, we collected 830 pleural fluid samples and reviewed 35 patients with pleural effusions undiagnosed after pleural biopsy at Yamaguchi...
4 CitationsSource
#1Shoko Matsui (University of Toyama)H-Index: 23
#2Hiroshi Yamamoto (Shinshu University)H-Index: 27
Last. Keishi KuboH-Index: 61
view all 6 authors...
Shoko Matsui, Hiroshi Yamamoto, Seijiro Minamoto, Yuko Waseda, Michiaki Mishima, Keishi Kubo Health Administration Center, University of Toyama, Gofuku 3190, Toyama-shi, Toyama 930-8555, Japan Shinshu University School of Medicine, Japan Osaka Prefectural Medical Center for Respiratory and Allergic Diseases, Japan Kanazawa University Graduate School of Medical Science, Japan Graduate School of Medicine, Kyoto University, Japan Nagano Prefectural Hospital Organization, Japan
45 CitationsSource
#1Yunyun Fei (Peking Union Medical College Hospital)H-Index: 19
#2Juhong ShiH-Index: 1
Last. Fengchun ZhangH-Index: 42
view all 13 authors...
To investigate clinical and radiological features of IgG4-related disease (IgG4-RD) patients with intrathoracic involvement. A prospective cohort study was performed and IgG4-RD patients were enrolled from January 2011 to March 2015 in Peking Union Medical College Hospital, in which the clinical and radiological characteristics of IgG4-RD patients with intrathoracic involvement were summarized. Out of total 248 cases with IgG4-RD, 87 cases had intrathoracic lesions, including 58 male cases and 2...
28 CitationsSource
#1Eun Kyong Goag (Yonsei University)H-Index: 3
#2Ji Eun Park (Yonsei University)H-Index: 5
Last. Kyung Soo Chung (Yonsei University)H-Index: 16
view all 12 authors...
IgG4-related disease is an immune-mediated fibro-inflammatory disease, characterized by lymphoplasmacytic infiltration composed of IgG4-positive plasma cells of various organs with elevated circulating levels of IgG4. This disease is now reported with increasing frequency and usually affects middle-aged men. Massive pleural effusion in children is an uncommon feature in IgG4-related disease. Here, we report a case of a 16-year-old male patient with extensive IgG4-related disease presenting with ...
8 CitationsSource
#1Eisuke KatoH-Index: 2
#2Noboru TakayanagiH-Index: 20
Last. Yutaka SugitaH-Index: 18
view all 6 authors...
Presently, 6 cases of IgG4-related pleuritis have been reported. We encountered a patient who developed chylothorax due to IgG4-related disease. To our knowledge, such patients have not been reported. This patient developed right-sided chylothorax and left-sided non-chylothorax lymphocyte-predominant pleuritis. Elevated serum and pleural IgG4 concentrations and histopathological analysis of pleural biopsy confirmed the diagnosis of IgG4-related pleuritis. Left-sided pleuritis improved with corti...
14 CitationsSource
#1Shoko Matsui (University of Toyama)H-Index: 23
#2Akira HebisawaH-Index: 20
Last. Takashi OguraH-Index: 16
view all 19 authors...
Background and objective Immunoglobulin G4 (IgG4)-related disease is a multi-organ disorder that can include the lungs. IgG4-related lung disease can present in various forms; the clinical, radiological and pathological features of patients with this disease have been assessed. Methods Forty-eight patients suspected of having IgG4-related lung disease, with a high serum concentration of IgG4 and abundant IgG4-positive plasma cell infiltration into the intrathoracic organs, were retrospectively e...
84 CitationsSource
#1John H. Stone (Harvard University)H-Index: 93
#2John K.C. ChanH-Index: 81
Last. Yoh ZenH-Index: 86
view all 6 authors...
IgG4-related disease (IgG4-RD) is a novel clinical disease entity characterized by an elevated serum IgG4 concentration and tumefaction or tissue infiltration by IgG4-positive plasma cells. IgG4-RD encompasses a wide variety of diseases, formerly diagnosed as Mikulicz's disease (MD), autoimmune pancreatitis (AIP), hypophysitis, Riedel thyroiditis, tubulointerstitial nephritis, prostatitis, retroperitoneal fibrosis, inflammatory aortic aneurysm and inflammatory pseudotumor. However, like a crow f...
365 CitationsSource
Cited By1
#1Masafumi ShimodaH-Index: 2
#2Yoshiaki TanakaH-Index: 14
Last. Ken OhtaH-Index: 53
view all 10 authors...
RATIONALE Levels of pleural fluid adenosine deaminase (ADA), a useful marker for the diagnosis of tuberculous pleurisy, are elevated in some reports of immunoglobulin G4 (IgG4)-related pleural effusion. We describe a patient with IgG4-related pleural effusion who exhibited a high concentration of ADA. Furthermore, we reviewed the literature to compare patients with IgG4-related pleural effusion and tuberculous pleurisy. PATIENT CONCERNS A 75-year-old male patient had dyspnea for 1 month with a l...