Immunoglobulin G4-related pleuritis – A case report

Gajewska, Marta; Rychwicka-Kielek, Beata A.; Sørensen, K H; Kubik, Magdalena; Hilberg, Ole; Bendstrup, Elisabeth

doi:10.1016/j.rmcr.2016.05.006

Cited by 13 publications

(11 citation statements)

References 18 publications

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“…Kondo and Uehara noted the accumulation of FDG in the pleura on PET-CT, which was not observed in our patient. Among previously reported cases of pleuritis, three did not have FDG accumulation (5,9,13), but two did (11,12). The serum IgG4 levels did not differ markedly between cases with and without positive PET scans.…”

Section: Discussionmentioning

confidence: 66%

“…Common radiologic findings in IgG4-related lung disease include mediastinal lymphadenopathy and thickening of the perilymphatic interstitium with or without subpleural and/or peribronchovascular consolidation (2). However, our patient exhibited only pleural effusion on chest CT; given the rarity of pleuritis as a form of IgG4-related disease (3)(4)(5)(6)(7)(8)(9)(10)(11)(12)(13), the diagnosis was not immediately obvious. Interestingly, the pleura was studded with numerous small, blister-like nodules, which was histopathologically consistent with IgG4related pleuritis.…”

Section: Discussionmentioning

confidence: 70%

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Thoracoscopic Findings in IgG4-related Pleuritis

et al. 2020

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A 46-year-old Japanese man was admitted to our hospital with a 1-year history of dyspnea and persistent right-dominant bilateral pleural effusions. Chest and abdominal computed tomography (CT) revealed no notable findings apart from the bilateral pleural effusions. 2-deoxy-2-[ 18 F]-fluoro-D-glucose (FDG) positron emission tomography-CT showed no accumulation of FDG in the thorax and abdomen. Thoracoscopy revealed numerous small (approximately 2-3 mm in size), blister-like nodules on the left parietal pleura extending from the lower third of the chest wall to the diaphragm. A pathological examination revealed lymphocyte and plasma cell infiltrates with increasing numbers of IgG4-positive plasma cells in the fibrotic pleura, indicating IgG4-related pleuritis.

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Section: Discussionmentioning

confidence: 66%

Section: Discussionmentioning

confidence: 70%

Thoracoscopic Findings in IgG4-related Pleuritis

et al. 2020

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“…Note that under a nontreatment state, the natural course in the two groups was different; no patient in the IgG4‐PL group improved completely, and half of the patients in the non‐IgG4‐PL group recovered spontaneously. Several reported cases and one patient (Case 7) of the IgG4‐PL group with massive PE responded well to steroids . However, despite steroid administration to one patient in the IgG4‐PL group (Case 6), PE and respiratory failure deteriorated.…”

Section: Discussionmentioning

confidence: 97%

“…Note that under a nontreatment state, the natural course in the two groups was different; no patient in the IgG4-PL group improved completely, and half of the patients in the non-IgG4-PL group steroids. 12,[16][17][18][19] However, despite steroid administration to one patient in the IgG4-PL group (Case 6), PE and respiratory failure deteriorated. We thought that the lack of treatment response in this patient could be caused by the inappropriate amount and/or length of the steroid management.…”

Section: Discussionmentioning

confidence: 99%

Clinicopathological features of immunoglobulin G4‐related pleural lesions and diagnostic utility of pleural effusion cytology

et al. 2018

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Objective Immunoglobulin (Ig)G4‐related disease is a recently described systemic immune‐mediated fibro‐inflammatory disease that frequently occurs in tumorous form. Herein, we elucidated the clinicopathological and cytological characteristics of IgG4‐related pleural lesions (PLs). Patients and methods Among 22 patients with fibro‐inflammatory PLs of idiopathic aetiology, eight cases were diagnosed as IgG4‐PL and the remaining 14 as non‐IgG4‐PL according to comprehensive diagnostic criteria for IgG4‐related disease. Cell block examination of pleural effusion (CBPE) was performed in five patients with IgG4‐PL and in six with non‐IgG4‐PL. Both groups were compared in terms of clinical presentation, laboratory data, histopathological features of resected pleura, and cytological features of pleural effusion (PE). Results PE was the most common (six patients, 75%) clinical presentation of IgG4‐PL. IgG4‐PL comparatively showed significantly more frequent concomitant allergic disease (P = .021), higher serum IgE levels (P = .012), higher adenosine deaminase levels in pleural fluid (P = .005), and rare spontaneous recovery without treatment (P = .046). The IgG4‐PL group was histologically characterised by thicker fibrous pleura, storiform fibrosis, and infiltration of regulatory T cells, eosinophils and basophils. Using CBPE, IgG4‐PL was cytologically distinct with numerous IgG4+ cells and eosinophils. The cytology of CBPE positively correlated with the histology of pleural tissue in the number of IgG4+ cells and eosinophils (R = .769 and .803, respectively). Conclusion IgG4‐PL frequently presents with PE and is histologically and cytologically characterised by abundant infiltration of IgG4+ cells and eosinophils. We believe that CBPE with immunohistochemistry/special staining could assist in the auxiliary diagnosis of IgG4‐PL.

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“…IgG4-related pleural lesions are reported to be steroidresponsive [26,32,62]. Considering B cell activation as a Receiver operating characteristic (ROC) analysis on diagnostic utility of immunoglobulin (Ig)G4 and j/k ratio for distinguishing patients between the IgG4 2 and IgG 1 groups.…”

Section: Discussionmentioning

confidence: 99%

Association of immunoglobulin G4 and free light chain with idiopathic pleural effusion

Murata

Aoe

Mimura‐Kimura

et al. 2017

Clinical and Experimental Immunology

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The cause of pleural effusion remains uncertain in approximately 15% of patients despite exhaustive evaluation. As recently described immunoglobulin (Ig)G4-related disease is a fibroinflammatory disorder that can affect various organs, including the lungs, we investigate whether idiopathic pleural effusion includes IgG4-associated etiology. Between 2000 and 2012, we collected 830 pleural fluid samples and reviewed 35 patients with pleural effusions undiagnosed after pleural biopsy at Yamaguchi-Ube Medical Center. Importantly, IgG4 immunostaining revealed infiltration of IgG4-positive plasma cells in the pleura of 12 patients (34%, IgG4 group). The median effusion IgG4 level was 41 mg/dl in the IgG4 group and 27 mg/dl in the IgG4 group (P < 0·01). The light and heavy chains of effusion IgG4 antibodies of patients in the IgG4 group were heterogeneous by two-dimensional electrophoresis, indicating the absence of clonality of the IgG4 antibodies. Interestingly, the κ light chains were more heterogeneous than the λ light chains. The measurement of the κ and λ free light chain (FLC) levels in the pleural fluids showed significantly different κ FLC levels (median: 28·0 versus 9·1 mg/dl, P < 0·01) and κ/λ ratios (median: 2·0 versus 1·2, P < 0·001) between the IgG4 and IgG4 groups. Furthermore, the κ/λ ratios were correlated with the IgG4 /IgG plasma cell ratios in the pleura of the IgG4 group. Taken together, these results demonstrate the involvement of IgG4 in certain idiopathic pleural effusions and provide insights into the diagnosis, pathogenesis and therapeutic opportunities of IgG4-associated pleural effusion.

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Immunoglobulin G4-related pleuritis – A case report

Cited by 13 publications

References 18 publications

Thoracoscopic Findings in IgG4-related Pleuritis

Thoracoscopic Findings in IgG4-related Pleuritis

Clinicopathological features of immunoglobulin G4‐related pleural lesions and diagnostic utility of pleural effusion cytology

Association of immunoglobulin G4 and free light chain with idiopathic pleural effusion

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