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IgG4-related disease can present as recurrent spontaneous hemothorax: a case report
BACKGROUND: Immunoglobulin G4-related disease (IgG4-RD) encompasses a group of immune-mediated disorders that are gaining increasing recognition. Pulmonary presentations are common, with four types of patterns been described on radiography, including solid nodular, bronchovascular, ground glass opac...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2019
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6359758/ https://www.ncbi.nlm.nih.gov/pubmed/30709387 http://dx.doi.org/10.1186/s12890-019-0785-y |
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author | Fan, Junping Feng, Ruie Hou, Xiaomeng Li, Ji Sun, Xuefeng Wang, Jinglan Shi, Juhong Wang, Mengzhao Xu, Yan |
author_facet | Fan, Junping Feng, Ruie Hou, Xiaomeng Li, Ji Sun, Xuefeng Wang, Jinglan Shi, Juhong Wang, Mengzhao Xu, Yan |
author_sort | Fan, Junping |
collection | PubMed |
description | BACKGROUND: Immunoglobulin G4-related disease (IgG4-RD) encompasses a group of immune-mediated disorders that are gaining increasing recognition. Pulmonary presentations are common, with four types of patterns been described on radiography, including solid nodular, bronchovascular, ground glass opacities, and alveolar interstitial. Pleural thickening and pleural effusion have also been reported. However, there have been no reports of IgG4-RD that presents as spontaneous hemothorax. CASE PRESENTATION: A 61-year-old Chinese woman experienced recurrent right-sided chest pain and transient syncope. A significant decrease in her hemoglobin level and thick bloody pleural fluid demonstrated spontaneous hemothorax. The elevated serum IgG4 and histopathological analysis of the right pleura and intercostal lymph node specimens all supported the diagnosis of IgG4-RD in this patient. Further diagnostic evaluation did not reveal other causes for spontaneous hemothorax. She received steroids and no recurrent bleeding event occurred during a follow-up period of more than 1 year. CONCLUSION: Recurrent spontaneous hemothorax can be a rare manifestation of IgG4-RD, with pleural involvement as the most probable mechanism. |
format | Online Article Text |
id | pubmed-6359758 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-63597582019-02-07 IgG4-related disease can present as recurrent spontaneous hemothorax: a case report Fan, Junping Feng, Ruie Hou, Xiaomeng Li, Ji Sun, Xuefeng Wang, Jinglan Shi, Juhong Wang, Mengzhao Xu, Yan BMC Pulm Med Case Report BACKGROUND: Immunoglobulin G4-related disease (IgG4-RD) encompasses a group of immune-mediated disorders that are gaining increasing recognition. Pulmonary presentations are common, with four types of patterns been described on radiography, including solid nodular, bronchovascular, ground glass opacities, and alveolar interstitial. Pleural thickening and pleural effusion have also been reported. However, there have been no reports of IgG4-RD that presents as spontaneous hemothorax. CASE PRESENTATION: A 61-year-old Chinese woman experienced recurrent right-sided chest pain and transient syncope. A significant decrease in her hemoglobin level and thick bloody pleural fluid demonstrated spontaneous hemothorax. The elevated serum IgG4 and histopathological analysis of the right pleura and intercostal lymph node specimens all supported the diagnosis of IgG4-RD in this patient. Further diagnostic evaluation did not reveal other causes for spontaneous hemothorax. She received steroids and no recurrent bleeding event occurred during a follow-up period of more than 1 year. CONCLUSION: Recurrent spontaneous hemothorax can be a rare manifestation of IgG4-RD, with pleural involvement as the most probable mechanism. BioMed Central 2019-02-01 /pmc/articles/PMC6359758/ /pubmed/30709387 http://dx.doi.org/10.1186/s12890-019-0785-y Text en © The Author(s). 2019 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. |
spellingShingle | Case Report Fan, Junping Feng, Ruie Hou, Xiaomeng Li, Ji Sun, Xuefeng Wang, Jinglan Shi, Juhong Wang, Mengzhao Xu, Yan IgG4-related disease can present as recurrent spontaneous hemothorax: a case report |
title | IgG4-related disease can present as recurrent spontaneous hemothorax: a case report |
title_full | IgG4-related disease can present as recurrent spontaneous hemothorax: a case report |
title_fullStr | IgG4-related disease can present as recurrent spontaneous hemothorax: a case report |
title_full_unstemmed | IgG4-related disease can present as recurrent spontaneous hemothorax: a case report |
title_short | IgG4-related disease can present as recurrent spontaneous hemothorax: a case report |
title_sort | igg4-related disease can present as recurrent spontaneous hemothorax: a case report |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6359758/ https://www.ncbi.nlm.nih.gov/pubmed/30709387 http://dx.doi.org/10.1186/s12890-019-0785-y |
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