Characteristics of pleural effusions in systemic lupus erythematosus: differential diagnosis of lupus pleuritis

Choi, Byeong Yoon; Yoon, M J; Shin, Kichul; Lee, Y J; Song, Yong-Wen

doi:10.1177/0961203314555171

Cited by 34 publications

(25 citation statements)

References 15 publications

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“…Case 1 presented as a posterior mediastinal mass, and tumors arising in visceral sites such as the small intestine and stomach have been reported. Tumor metastases (synchronous and metachronous) were observed in both cases and have been similarly described in prior reports, with the lung as a common site: Italiano et al reported metastases in 8 of 15 patients (53%), and all 4 patients reported by Choi et al developed metastases (including 3 at the time of initial diagnosis). Data are somewhat limited regarding outcome but indicate an aggressive course; Choi et al found that all 4 patients in their series died of disease within 17 months despite all having undergone surgical resection with receipt of neoadjuvant chemotherapy and/or radiotherapy .…”

Section: Discussionsupporting

confidence: 82%

“…CIC‐DUX4 sarcoma appears to affect younger patients, with a median age of diagnosis (approximately 26 years) during the third decade of life, with relatively equal distribution between sexes. However, a wide age range from 6 to 62 years has been reported, and thus older age (as in the patient in case 2) should not be a diagnostic deterrent. Tumors most frequently arise in the somatic soft tissue of the extremities and trunk .…”

Section: Discussionmentioning

confidence: 99%

“…However, a wide age range from 6 to 62 years has been reported, and thus older age (as in the patient in case 2) should not be a diagnostic deterrent. Tumors most frequently arise in the somatic soft tissue of the extremities and trunk . Case 1 presented as a posterior mediastinal mass, and tumors arising in visceral sites such as the small intestine and stomach have been reported.…”

Section: Discussionmentioning

confidence: 99%

“…CIC‐DUX4 fusion has been identified in up to two‐thirds of EWSR1‐ negative round cell sarcomas. The translocation involves the CIC locus on 19q13 with 1 of 2 DUX4 loci (either 4q35 or 10q26.3) . Published reports and series have characterized tumors (approximately 40 in total) as predominantly affecting pediatric and young adult patients, and describe the histopathologic and immunophenotypic features in surgical resection specimens.…”

Section: Introductionmentioning

confidence: 99%

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Round cell sarcoma with CIC‐DUX4 gene fusion: Discussion of the distinctive cytomorphologic, immunohistochemical, and molecular features in the differential diagnosis of round cell tumors

Chebib

2016

Cancer Cytopathology

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BACKGROUND: Undifferentiated round cell sarcomas are a heterogeneous group, and include tumors that resemble the Ewing sarcoma family. Although a subset defined by recurrent CIC-DUX4 gene fusion has been recently characterized, data regarding the cytomorphologic features are currently limited. Two recent fine-needle aspiration (FNA) cases prompted review of the spectrum of round cell tumors in the differential diagnosis to determine distinctive diagnostic features. METHODS: Two genetically confirmed FNA cases were identified. Cytomorphologic features were evaluated on FNA smears and hematoxylin and eosin-stained cell block and concurrent needle biopsy sections, and immunohistochemical studies performed on cell block and biopsy sections were reviewed. RESULTS: The 2 patients were a 24-year-old man with a posterior mediastinal mass and a 69-year-old woman with a gluteal mass. FNA smears were cellular with tumor cells present in large groups and singly dispersed. Tumor cells had large, round-to-ovoid, hyperchromatic nuclei with irregular membranes, frequent large nucleoli, and a moderate amount of vacuolated cytoplasm. Both cases demonstrated necrosis, and one case had prominent myxoid stroma. Immunohistochemistry demonstrated focal-to-multifocal CD99 positivity and diffuse nuclear staining for WT1; staining for cytokeratin, desmin, S-100, CD34, CD45, and TdT were negative. Fluorescence in situ hybridization demonstrated CIC-DUX4 fusion in both cases. CONCLUSIONS: CIC-DUX4 round cell sarcoma differs from Ewing sarcoma in that it has more atypical cytologic features and lacks the diffuse membranous CD99 staining pattern characteristic of Ewing sarcoma. The differential diagnosis is broad, and requires the judicious use of ancillary studies.Focal-to-multifocal CD99 immunoreactivity and diffuse nuclear WT1 positivity is characteristic of CIC-DUX4 sarcoma, and should prompt molecular testing. Cancer Cytopathol 2016;124:350-61.

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Section: Discussionsupporting

confidence: 82%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Round cell sarcoma with CIC‐DUX4 gene fusion: Discussion of the distinctive cytomorphologic, immunohistochemical, and molecular features in the differential diagnosis of round cell tumors

Chebib

2016

Cancer Cytopathology

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“…However, a variety of immunopathogenic mechanisms are involved, including TH1 and TH17 cell-mediated immunity and acute inflammation 2. Serositis is a frequent manifestation in SLE and can be the presenting feature 3. The pleura is involved more commonly in SLE than in any other connective tissue disease.…”

Section: Introductionmentioning

confidence: 99%

Successful treatment of recurrent pleural and pericardial effusions with tocilizumab in a patient with systemic lupus erythematous

Ocampo¹,

Haaland

Legault

et al. 2016

BMJ Case Reports

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A 22-year-old Caucasian man presented to hospital with pleuritic chest pain. He had had a history of a sun-sensitive rash a year prior. Workup revealed normal cardiac enzymes and chest X-ray. However, electrocardiogram revealed ST elevation and PR depression, and echocardiogram revealed a slight pericardial effusion without other findings. A diagnosis of pericarditis was made. Subsequently, he was found to be positive for antinuclear antibodies (ANAs), as well as antibodies to SSA, SSB and double-stranded DNA; C3 was low, and C4 was undetectable. A diagnosis of systemic lupus erythematosus was made. The patient initially responded to high-dose ibuprofen. One month later, he developed a new pericardial effusion, this time with concomitant massive left-sided pleural effusion, requiring three separate thoracenteses draining a total of 6 L of pleural fluid. The recurrent effusion failed to respond to high-dose corticosteroid treatment. Owing to the severity and rapidity of the recurrence of pleural and pericardial effusion, intravenous tocilizumab was administered. The patient had excellent clinical and radiographic improvement. This case shows that tocilizumab may have a role in the treatment of intractable pleuropericardial effusion and other forms of lupus-associated serositis.

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Diagnosis of systemic lupus erythematosus in a dog with an atypical presentation of pleuritis

Oh,

Melega,

Bussey

et al. 2023

Vet Record Case Reports

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A 3‐year‐old labradoodle presented with prior history of generalised seizures and had recently developed acute pyrexia and shifting leg lameness, with joint effusions in several joints. The dog also later developed pleuritis with an exudative pleural effusion. Cytological findings for all joint effusions were consistent with immune‐mediated polyarthritis. Suspected lupus erythematosus cells were seen in the joints and pleural fluid, which were confirmed with Feulgen staining. Together with a positive antinuclear antibody titre, the dog was diagnosed with systemic lupus erythematosus, which responded to immunosuppressive doses of prednisolone. This case report describes the insidious nature of systemic lupus erythematosus, a rare disease that can imitate many different conditions. It is also the first reported case of lupus erythematosus cells in pleural effusion in veterinary medicine, highlighting the importance of lupus erythematosus cells as a valuable diagnostic aid to further support the diagnosis of systemic lupus erythematosus and enable early treatment to be initiated.

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Characteristics of pleural effusions in systemic lupus erythematosus: differential diagnosis of lupus pleuritis

Cited by 34 publications

References 15 publications

Round cell sarcoma with CIC‐DUX4 gene fusion: Discussion of the distinctive cytomorphologic, immunohistochemical, and molecular features in the differential diagnosis of round cell tumors

Round cell sarcoma with CIC‐DUX4 gene fusion: Discussion of the distinctive cytomorphologic, immunohistochemical, and molecular features in the differential diagnosis of round cell tumors

Successful treatment of recurrent pleural and pericardial effusions with tocilizumab in a patient with systemic lupus erythematous

Diagnosis of systemic lupus erythematosus in a dog with an atypical presentation of pleuritis

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