Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review

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...

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Autores principales: Sakata, Komei, Kikuchi, Jun, Emoto, Katsura, Kotaki, Tomomi, Ota, Yuichiro, Nishina, Naoshi, Hanaoka, Hironari, Otomo, Kotaro, Suzuki, Katsuya, Kaneko, Yuko, Takeuchi, Tsutomu
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Japanese Society of Internal Medicine 2021
Materias:
Case Report
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8313917/
https://www.ncbi.nlm.nih.gov/pubmed/33518567
http://dx.doi.org/10.2169/internalmedicine.6313-20
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author Sakata, Komei
Kikuchi, Jun
Emoto, Katsura
Kotaki, Tomomi
Ota, Yuichiro
Nishina, Naoshi
Hanaoka, Hironari
Otomo, Kotaro
Suzuki, Katsuya
Kaneko, Yuko
Takeuchi, Tsutomu
author_facet Sakata, Komei
Kikuchi, Jun
Emoto, Katsura
Kotaki, Tomomi
Ota, Yuichiro
Nishina, Naoshi
Hanaoka, Hironari
Otomo, Kotaro
Suzuki, Katsuya
Kaneko, Yuko
Takeuchi, Tsutomu
author_sort Sakata, Komei
collection PubMed
description 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.
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spelling pubmed-83139172021-08-11 Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review Sakata, Komei Kikuchi, Jun Emoto, Katsura Kotaki, Tomomi Ota, Yuichiro Nishina, Naoshi Hanaoka, Hironari Otomo, Kotaro Suzuki, Katsuya Kaneko, Yuko Takeuchi, Tsutomu Intern Med Case Report 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. The Japanese Society of Internal Medicine 2021-02-01 2021-07-01 /pmc/articles/PMC8313917/ /pubmed/33518567 http://dx.doi.org/10.2169/internalmedicine.6313-20 Text en Copyright © 2021 by The Japanese Society of Internal Medicine https://creativecommons.org/licenses/by-nc-nd/4.0/The Internal Medicine is an Open Access journal distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit (https://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Sakata, Komei
Kikuchi, Jun
Emoto, Katsura
Kotaki, Tomomi
Ota, Yuichiro
Nishina, Naoshi
Hanaoka, Hironari
Otomo, Kotaro
Suzuki, Katsuya
Kaneko, Yuko
Takeuchi, Tsutomu
Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review
title Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review
title_full Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review
title_fullStr Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review
title_full_unstemmed Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review
title_short Refractory IgG4-related Pleural Disease with Chylothorax: A Case Report and Literature Review
title_sort refractory igg4-related pleural disease with chylothorax: a case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8313917/
https://www.ncbi.nlm.nih.gov/pubmed/33518567
http://dx.doi.org/10.2169/internalmedicine.6313-20
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