Mechanisms of Pleural Involvement in Orphan Diseases

Anevlavis, Stavros; Τzouvelekis, Αrgyris; Bouros, Demosthenes

doi:10.1159/000335128

Cited by 12 publications

(12 citation statements)

References 94 publications

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“…Suggested mechanisms are presumably similar to those of other infiltrative diseases. Involvement of the pleura may lead to increased capillary permeability with minimal pleural space inflammation [10]. In a recent study of patients with sarcoidosis, pleural effusion was diagnosed in five out of 181 cases by using chest ultrasound, whereas a biopsy-proven diagnosis was made in only two cases [11].…”

Section: Discussionmentioning

confidence: 99%

Sarcoidosis in a 65-year-old woman presenting with a lung mass and pericardial effusion: a case report

Margaritopoulos¹,

Proklou²,

Lagoudaki³

et al. 2012

J Med Case Reports

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IntroductionSarcoidosis is a multi-systemic disorder of unknown origin and most commonly affects the lungs. Diagnosis relies on the presence of non-caseating granulomas on histologic specimens. In high-resolution computed tomography, the most characteristic findings are peribronchovascular thickening, perilymphatic nodular distribution, and bilateral hilar adenopathy. Confluent nodular opacities or large masses are rare manifestations of the disease. It is well recognized that sarcoidosis can mimic infectious, malignant, and granulomatous conditions. Here, we report a case with a high initial index of suspicion for lung malignancy in terms of clinical, lung imaging, and endoscopic findings.Case presentationA 65-year-old Caucasian woman, lifelong non-smoker with an unremarkable medical history, presented with a 10-month history of progressive breathlessness, dry cough, fatigue, arthralgias, and mild weight loss. The only significant clinical finding was bilateral enlargement of auxiliary lymph nodes. High-resolution computed tomography revealed a soft tissue density mass at the right hilum which was surrounding and narrowing airways and vascular components, nodules with vascular distribution, enlarged mediastinal lymph nodes, and pericardial effusion. Our patient underwent a bronchoscopy, which revealed the presence of submucosal infiltration and narrowing of the right upper bronchus. Endobronchial biopsies showed non-caseating granulomas. As local sarcoid reactions with non-caseating granulomas can be observed near tumors, our patient underwent video-assisted thoracoscopy and surgical removal of an auxiliary lymph node, both of which confirmed the presence of non-caseating granulomas and the diagnosis of sarcoidosis. She was treated with steroids with improvement of clinical and imaging findings. However, while on a maintenance dose, she presented with a pleural effusion, which, after the diagnostic work-up, proved to be sarcoidosis-related. Treatment with initially high doses of steroids plus a steroid-sparing agent led to resolution of the effusion.ConclusionsWe report a case with a high initial index of suspicion for lung malignancy. Clinicians should always be aware that sarcoidosis enters the differential diagnosis of patients presenting with a lung mass that encases and narrows bronchial and vascular structures with associated pericardial effusion. Rarely, pleural effusion can be the presenting symptom of disease relapse despite maintenance treatment.

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Section: Discussionmentioning

confidence: 99%

Sarcoidosis in a 65-year-old woman presenting with a lung mass and pericardial effusion: a case report

Margaritopoulos¹,

Proklou²,

Lagoudaki³

et al. 2012

J Med Case Reports

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“…Thoracoscopy in this patient population is non‐specific, yet occasionally it can reveal nodules on the visceral pleura and immunofluorescence of biopsy samples of these nodules revealed immunoglobulin deposits. The definitive therapy includes immune‐suppression, but for the resistant cases, pleurodesis might be attempted …”

Section: Congestive Heart Failure Hydrothoraxmentioning

confidence: 99%

“…Cytology may occasionally show multinucleated giant cells. Minority of sarcoid effusions are chylothorax caused by thoracic and abdominal lymphatic duct obstruction and hemothorax …”

Section: Congestive Heart Failure Hydrothoraxmentioning

confidence: 99%

Is there any role for thoracoscopy in the diagnosis of benign pleural effusions

Anevlavis

Varga

Nam

et al. 2019

Clinical Respiratory J

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Thoracoscopy in the endoscopy suite, has a high diagnostic yield of undiagnosed pleural effusions with minimal and mild complications. Whereas relatively minimal invasive techniques, such as thoracentesis, image-guided pleural biopsy or blind pleural biopsy, can yield sufficient cell or tissue material to establish the diagnosis of the underlying condition, more definite invasive diagnostic and therapeutic procedure, such as thoracoscopy, may be required for accurate sampling and diagnosis, and further provide real-time treatment options in same procedure. If thoracoscopy is considered the gold standard for the diagnosis is a fact in case. The current review aims to provide informations on thoracoscopy indications in benign pleural diseases according to up to date publications. K E Y W O R D Sbenign pleural effusion, empyema medical thoracoscopy, pleural infections, tuberculous pleural effusion 74 | ANEVLAVIS Et AL.

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“…Less than 5% of patients with RA may experience in life a pleural effusion, mainly aged males with subcutaneous rheumatoid nodules [27,46,47] ( fig. 2 a, b).…”

Section: Pleural Diseasementioning

confidence: 99%

“…(1) elongated macrophages, (2) round multinucleated macrophages and (3) background of granular necrotic debris [50] . Ragocytes, IgG and/or RF inclusions in multinucleated macrophages may be nonspecific [47] . If surgical biopsy becomes necessary, rheumatoid nodules may be observed at the level of the pleural surfaces ( fig.…”

Section: Pleural Diseasementioning

confidence: 99%

Investigation of Lung Involvement in Connective Tissue Disorders

Papiris¹,

Manali²,

Kolilekas³

et al. 2015

Respiration

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Lung involvement in connective tissue disorders (CTDs) may present as pleomorphic since any lung compartment may be involved such as airways, exocrine secretory and alveolar epithelia, interstitial lung structure, pulmonary vasculature and pleura as well as, in specific disorders, several tissues of the thoracoabdominal ventilator pump. Any combination of the above anatomic structures may be involved concomitantly although some specific combinations may include a determinant of rheumatic disorders. The diagnosis of a specific CTD requires the fulfilment of clearly defined clinical and laboratory criteria including in most cases positivity for autoantibodies, mostly specific serologic combinations. In this setting, serologic investigation targets mainly, although not exclusively, the detection of antinuclear antibodies. A specific serologic positivity or a combination of autoantibodies constitutes not only a diagnostic criterion for a specific CTD, but may also characterize the pattern of respiratory manifestation in a determinant rheumatic disorder. Therefore, the investigation of lung involvement in CTDs requires adequate skills in the ambit of a multidisciplinary approach and an extended spectrum of diagnostic tools and modalities able to detect both early clinical clues and serologic conversion as well as any pathophysiologic alteration that regards the complexity of respiratory functional status. Although many patients with CTDs suffer from a ‘vicious' combination of lung involvement, lung drug toxicity and infections related to the above two as well as to the ‘mater' disease, for space reasons this review will focus on the established lung manifestations that regard the 7 major CTDs.

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Mechanisms of Pleural Involvement in Orphan Diseases

Cited by 12 publications

References 94 publications

Sarcoidosis in a 65-year-old woman presenting with a lung mass and pericardial effusion: a case report

Sarcoidosis in a 65-year-old woman presenting with a lung mass and pericardial effusion: a case report

Is there any role for thoracoscopy in the diagnosis of benign pleural effusions

Investigation of Lung Involvement in Connective Tissue Disorders

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