IgG4-related pleural disease with aortitis and submandibular glands involvement successfully treated with corticosteroid: case-based review

被引：8

作者：

Okamoto, Shota ^{[1
]}

Tsuboi, Hiroto ^{[1
]}

Sato, Ryota ^{[1
]}

Terasaki, Mayu ^{[1
]}

Terasaki, Toshihiko ^{[1
]}

Toko, Hirofumi ^{[1
]}

Shimizu, Masaru ^{[1
]}

Honda, Fumika ^{[1
]}

Yagishita, Mizuki ^{[1
]}

Ohyama, Ayako ^{[1
]}

Kurata, Izumi ^{[1
]}

Abe, Saori ^{[1
]}

Takahashi, Hiroyuki ^{[1
]}

Osada, Atsumu ^{[1
]}

Hagiwara, Shinya ^{[1
]}

Kondo, Yuya ^{[1
]}

Matsumoto, Isao ^{[1
]}

Sumida, Takayuki ^{[1
]}

机构：

[1] Univ Tsukuba, Fac Med, Dept Internal Med, 1-1-1 Tennodai, Tsukuba, Ibaraki 3058575, Japan

来源：

RHEUMATOLOGY INTERNATIONAL | 2020年 / 40卷 / 10期

基金：

日本学术振兴会;

关键词：

IgG4-related disease; Pleural disease; Periaortitis; Submandibular gland; DIAGNOSTIC-CRITERIA; PLEURITIS; LUNG; STATEMENT;

D O I：

10.1007/s00296-020-04555-y

中图分类号：

R5 [内科学];

学科分类号：

1002 ; 100201 ;

摘要：

IgG4-related disease (IgG4-RD) is a fibro-inflammatory condition characterized by increased serum IgG4 level, infiltration of lymphocytes and IgG4-positive (IgG4(+)) plasma cells and fibrosis. It can occur in almost all organs, commonly affecting the pancreas, biliary tract, salivary and lacrimal glands and kidneys. However, reports of IgG4-RD accompanied by pathologically confirmed, IgG4-related pleural disease are scarce. Here, we present a case of a 64-year-old man with suspected malignant pleural mesothelioma based on imaging findings but finally diagnosed with IgG4-RD (including pleuritis, periaortitis and bilateral submandibular gland enlargement) based on a high serum IgG4 level and pleural histopathological findings such as lymphoplasmacytic infiltration including IgG4(+) plasma cells and fibrosis. Systemic corticosteroid therapy was effective at reducing serum IgG4, improving bilateral submandibular gland enlargement, and regressing pleural thickening and periaortic soft tissue. We also discuss clinical characteristics and pleural pathological features of previously reported cases with IgG4-related pleural disease based on a comprehensive literature review. Our case of IgG4-RD with pleura, aorta and submandibular gland involvement, pathologically confirmed by pleural specimen might be unique and very rare.

引用

页码：1725 / 1732

页数：8

共 35 条

[21] Immunoglobulin G4-related disease with fibroinflammatory lesions in the pleura, bile ducts and pericardium [J].