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Review Case Reports
Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review.
- Komei Sakata, Jun Kikuchi, Katsura Emoto, Tomomi Kotaki, Yuichiro Ota, Naoshi Nishina, Hironari Hanaoka, Kotaro Otomo, Katsuya Suzuki, Yuko Kaneko, and Tsutomu Takeuchi.
- Division of Rheumatology, Department of Internal Medicine, Keio University School of Medicine, Japan.
- Intern. Med. 2021 Jul 1; 60 (13): 2135-2143.
AbstractWe herein report a rare case of a 66-year-old man with refractory chylothorax. Although he had been treated with moderate doses of prednisolone (PSL) on suspicion of pleuritis with Sjögren syndrome, the pleural effusion expanded after the reduction of PSL. Further workup including histopathological examinations of pleura led to the diagnosis of IgG4-RD with bilateral chylothorax without any leakage from the thoracic duct. Combination therapy with high-dose PSL plus rituximab successfully decreased the pleural effusion. This is a very rare case of IgG4-related pleuritis with chylothorax and the first report of its successful treatment with rituximab.
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