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Case Reports
. 2018 Aug 1;57(15):2251-2257.
doi: 10.2169/internalmedicine.0387-17. Epub 2018 Mar 9.

IgG4-related Pleuritis with Elevated Adenosine Deaminase in Pleural Effusion

Atsushi Nagayasu  1 , Satoshi Kubo  1 , Kazuhisa Nakano  1 , Shingo Nakayamada  1 , Shigeru Iwata  1 , Ippei Miyagawa  1 , Shunsuke Fukuyo  1 , Kazuyoshi Saito  2 , Yoshiya Tanaka  1
Affiliations
Case Reports

IgG4-related Pleuritis with Elevated Adenosine Deaminase in Pleural Effusion

Atsushi Nagayasu et al. Intern Med. .

Abstract

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-related pleuritis and treated with the patient with glucocorticoid therapy. The ADA levels in pleural effusion can increase in IgG4-related pleuritis, and it is therefore important to perform a pleural biopsy.

Keywords: IgG4-related disease with pleuritis; adenosine deaminase; pleural biopsy; pleural effusion; tuberculous pleurisy.

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Figures

Figure 1.
Figure 1.
Changes in the level of pleural effusion after treatment. A chest X-ray (A) and contrast-enhanced CT scan (B) on admission day showed massive bilateral pleural effusion. Contrast-enhanced chest CT of the neck to pelvis also demonstrated subpleural ground-glass opacity in both lungs, consolidation of the left lobe and mild mediastinal lymphadenopathy. No mass lesions or pleural thickening were found. After 4 weeks of treatment with systemic steroids, a chest X-ray (C) and CT scan (D) showed markedly decreased levels of bilateral pleural effusion.
Figure 2.
Figure 2.
Histopathological evaluation of the pleura and pleural effusion. A pleural biopsy showed the presence of dense lymphoplasmacytic infiltrate [(A): Hematoxylin and Eosin (H&E) staining, ×40; (B): H&E staining, ×100]. Immunohistochemically, many IgG4-poisitive plasma cells are identified (IgG4-positive plasma cells >50/HPF, IgG4+/IgG+cell ration <40%). [(C): immunohistochemical staining for IgG, ×400; (D): immunohistochemical staining for IgG4, ×400]. (E) Ziehl-Neelsen staining identified no acid-fast bacteria.
Figure 3.
Figure 3.
Clinical course. The administration of PSL resulted in an improvement of dyspnea and a decrease in the serum IgG4 level. IgG: immunoglobulin G (solid line), IgG4: immunoglobulin G4 (dotted line). PSL: prednisolone
See this image and copyright information in PMC

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