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

doi:10.2169/internalmedicine.6313-20

Cited by 4 publications

(3 citation statements)

References 53 publications

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“…In the cases analyzed ( Table 1 ), corticosteroids were used as initial treatment, resulting in symptom improvement in most patients. Immunosuppressants or rituximab (RTX), a monoclonal antibody targeting CD20, may be considered in cases of recurrence, hormonal- related adverse reactions, or multiorgan involvement ( 11 , 16 ). Surgical treatment may be effective for IgG4-PL patients who experience recurrent pleural effusion after GC combined with immunosuppressant therapy.…”

Section: Discussionmentioning

confidence: 99%

IgG4-related diseases involving pleura: a case report and literature review

Zuo,

Liu,

Guo

et al. 2023

Front. Med.

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Immunoglobulin G4-related disease (IgG4-RD) is a systemic fibro-inflammatory disease with the potential to involve virtually all organs, including the pancreas, kidneys, lungs, and pleura, amongst others. IgG4-RD pleural involvement may cause diverse complications such as pleural effusion, pleural thickening, pleural nodules, and additional lesions, which can be presented in many clinical diseases. However, isolated cases of pleurisy are still rare in IgG4-RD. We report a 72-year-old patient who was admitted to our hospital with cough, expectoration, and fatigue. He had a right-sided pleural effusion, and the tissue evaluation of the pleural biopsy by medical thoracoscopy met the diagnostic criteria of IgG4-RD. His serum IgG4 levels were elevated and he was finally diagnosed with IgG4-RD pleural involvement. He was subsequently started on prednisone 40 mg daily and his pleural effusion was almost disappeared 2 weeks later. This paper reported a case of IgG4-RD who had exclusive involvement of the pleura and highlighted the significance of considering IgG4-RD as a potential diagnosis in patients with unexplained pleural effusion.

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Section: Discussionmentioning

confidence: 99%

IgG4-related diseases involving pleura: a case report and literature review

Zuo,

Liu,

Guo

et al. 2023

Front. Med.

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“…Metachronous pathology is a characteristic phenomenon that may occur in multiple organs in patients with IgG4‐RD. Bilateral IgG4‐associated pleuritis has been reported in 8 cases, 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 and metachronous IgG4‐related pleuritis has not been reported.…”

Section: Discussionmentioning

confidence: 99%

Metachronous bilateral immunoglobulin G4‐related pleuritis: A case report and literature review

Tanabe

Homma

Matsui

et al. 2023

Respirology Case Reports

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Immunoglobulin G4‐related disease (IgG4‐RD) is a newly recognized disease, and therefore its clinical features are not yet fully understood. Here, we describe a surgical case of metachronous bilateral IgG4‐related pleuritis and postoperative chylothorax. This case could provide key insights into the pathology of IgG4‐RD from a surgical perspective. We present a 70‐year‐old woman who had a right pleural mass. Video‐assisted thoracoscopic pleural mass resection was performed, and the patient was diagnosed with right‐sided IgG4‐related pleuritis. Two years later, she was also diagnosed with left‐sided IgG4‐related pleuritis. We suspected the presence of IgG4‐positive plasma cell infiltration. Additionally, she experienced a complicated postoperative chylothorax on the left side. It is important to consider the altered course of lymphatic vessels when extensively removing the pleura near the right thoracic duct. The occurrence of metachronous bilateral IgG4‐associated pleuritis has not been previously reported, making this case particularly significant for understanding the pathology of IgG4‐RD from a surgical standpoint.

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“…Pleural involvement is also common, manifesting as chronic or recurring pleural effusions with characteristic pleural thickening (40, 41) (Figure 1b). Effusions are generally exudative, though chylous effusions have been described as well (41)(42)(43). Pericardial involvement in IgG4-RD is common and can lead to pericardial effusions, pericardial thickening, and constrictive pericarditis.…”

Section: Chestmentioning

confidence: 99%

Clinical Perspectives on IgG4-Related Disease and Its Classification

Katz¹,

Stone²

2022

Annu. Rev. Med.

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Immunoglobulin G4–related disease (IgG4-RD) is a systemic immune-mediated fibroinflammatory disease. Since its discovery nearly two decades ago, our understanding of its pathophysiology and clinical manifestations has grown substantially. Early diagnosis and treatment of this elusive disease can prevent substantial organ damage from end-stage fibrosis, emphasizing the need for prompt recognition and accurate characterization of IgG4-RD. The classification criteria endorsed by the American College of Rheumatology and the European Alliance of Associations for Rheumatology in 2019 provide a framework for establishing the diagnosis in the clinical setting. This process involves recognizing the typical manifestations of the disease and incorporating clinical, radiological, serological, and histopathological information as well as excluding disease mimickers. Glucocorticoids and rituximab are effective at inducing remission in IgG4-RD in most patients, but the optimal approach to long-term management of IgG4-RD remains an area of active clinical research.

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

Cited by 4 publications

References 53 publications

IgG4-related diseases involving pleura: a case report and literature review

IgG4-related diseases involving pleura: a case report and literature review

Metachronous bilateral immunoglobulin G4‐related pleuritis: A case report and literature review

Clinical Perspectives on IgG4-Related Disease and Its Classification

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