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; Tsutomu Takeuchi

doi:10.2169/internalmedicine.6313-20

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, Tsutomu Takeuchi

Research output: Contribution to journal › Article › peer-review

2 Citations (Scopus)

Abstract

We 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.

Original language	English
Pages (from-to)	2135-2143
Number of pages	9
Journal	Internal Medicine
Volume	60
Issue number	13
DOIs	https://doi.org/10.2169/internalmedicine.6313-20
Publication status	Published - 2021

Keywords

Chylothorax
IgG4-related disease
Pleuritis
Rituximab

ASJC Scopus subject areas

Internal Medicine

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10.2169/internalmedicine.6313-20

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title = "Refractory IgG4-related pleural disease with chylothorax: A case report and literature review",

abstract = "We 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 Sj{\"o}gren 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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author = "Komei Sakata and Jun Kikuchi and Katsura Emoto and Tomomi Kotaki and Yuichiro Ota and Naoshi Nishina and Hironari Hanaoka and Kotaro Otomo and Katsuya Suzuki and Yuko Kaneko and Tsutomu Takeuchi",

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T1 - Refractory IgG4-related pleural disease with chylothorax

T2 - A case report and literature review

AU - Sakata, Komei

AU - Kikuchi, Jun

AU - Emoto, Katsura

AU - Kotaki, Tomomi

AU - Ota, Yuichiro

AU - Nishina, Naoshi

AU - Hanaoka, Hironari

AU - Otomo, Kotaro

AU - Suzuki, Katsuya

AU - Kaneko, Yuko

AU - Takeuchi, Tsutomu

PY - 2021

Y1 - 2021

N2 - We 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.

AB - We 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.

KW - Chylothorax

KW - IgG4-related disease

KW - Pleuritis

KW - Rituximab

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JO - Internal Medicine

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ER -

Refractory IgG4-related pleural disease with chylothorax: A case report and literature review

Abstract

Keywords

ASJC Scopus subject areas

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