Chylous Pleural Effusion Associated With Primary Lymphedema and Lymphangioma-like Malformations

Bresser, Paul; Kromhout, J. G.; Reekers, Jim A.; Verhage, T.L.

doi:10.1378/chest.103.6.1916

Cited by 10 publications

(8 citation statements)

References 14 publications

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“…Non-traumatic chylothorax secondary to filariasis, postinflammatory thoracic duct obstruction, idiopathic lymphangiectasis with lymph node hypoplasia, malignancy, subclavian vein thrombosis or lymphangioma are much less common [5,7,8]. Bilateral chylous effusions do occur and are associated with a poor prognosis [1].…”

Section: Discussionmentioning

confidence: 99%

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Pseudochylothorax in pleural effusion due to coronary artery bypass surgery

García-Pachón

Fernandez²,

López-Azorín

et al. 1999

Eur Respir J

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This study describes a 64-yr-old male with a chronic left pleural effusion following a coronary artery bypass 3 yrs earlier. On thoracocentesis, turbid fluid was obtained with crystals of cholesterol on microscopic examination, establishing the diagnosis of pseudochylothorax. The pleural fluid cholesterol level was 207 mg. dL-1 (5.36 mmol. L-1). This is the first report of pseudochylothorax in a chronic pleural effusion due to coronary artery bypass surgery. Eur Respir J 1999; 13: 1487±1488. Pseudochylothorax is a pleural effusion with a very high content of cholesterol [1]. It can occur when fluid has been present in the pleural space for a long time especially with fibrotic pleura [1, 2]. The three most common causes of pseudochylothorax are tuberculous pleural effusion, therapeutic pneumothorax, and chronic rheumatoid pleurisy [1±4], but it can develop with other diseases which cause extensive fibrosis of the pleura [1]. Chronic pleural ef-fusion and pleural thickening may occur following coronary artery bypass, but this procedure has not previously been described as a cause of pseudochylothorax. Case report A 64-year-old male with a past medical history of my-ocardial infarction and coronary artery bypass surgery was admitted to the authors' hospital because of a decompen-sation of congestive heart failure which resolved in a few days following conventional therapy. Chest radiographs during the admission showed an unchanging pleural effu-sion that occupied more than one-third of the left hemi-thorax. The patient was aware that he had a chronic pleural effusion after having undergone coronary artery bypass 3 yrs earlier at another hospital. The radiograph prior to surgery was normal and he had no history of previous pleural disease, tuberculosis, or rheumatoid arthritis. The cardiac surgeon followed the patient for 1 yr and informed him that he had a left pleural effusion which had not changed during this period. At that time, no thoraco-centesis or other diagnostic tests were performed. On admission to our hospital, a chest computed tomographic scan showed the large left pleural effusion with thickened pleura and a small right pleural effusion. Thoracocentesis was performed and turbid fluid was obtained. Examination for malignant cells was negative, as were stains and cultures for bacteria, mycobacteria and fungi. Pleural fluid analysis showed an exudative effusion with a cholesterol level of 207 mg. dL-1 (5.36 mmol. L-1) and triglycerides 81 mg. dL-1 (0.90 mmol. L-1). Crystals of cholesterol were seen on microscopic examination, confirming the diagnosis of pseu-dochylothorax. The patient declined to undergo pleural decortication. Discussion

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Section: Discussionmentioning

confidence: 99%

“…Oedema of the lower extremities and body wall can arise from increased capillary permeability or obstruction of the lymphatic channels and an imbalance between capillary filtration and lymph drainage [5,6]. Chronic lymphoedema in association with chylous effusions rarely occurs.…”

mentioning

confidence: 99%

Pseudochylothorax in pleural effusion due to coronary artery bypass surgery

García-Pachón

Fernandez²,

López-Azorín

et al. 1999

Eur Respir J

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“…Sometimes massive pleural effusion and progressive pulmonary infiltration cause respiratory failure [15]. Pleural effusions are often chylous, due to spontaneous rupture of diseased lymph vessels within the lymphangiomas [26], and patients may have associated chyloptysis [29], haemoptysis [6,30,31] or chylopericardium [32]. In some case reports, chylous ascites, protein wasting enteropathy, peripheral lymphoedema, hemihyperplasia and lymphopenia have been also described [1] (figure 3).…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Thoracic involvement in generalised lymphatic anomaly (or lymphangiomatosis)

2016

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Generalised lymphatic anomaly (GLA), also known as lymphangiomatosis, is a rare disease caused by congenital abnormalities of lymphatic development. It usually presents in childhood but can also be diagnosed in adults. GLA encompasses a wide spectrum of clinical manifestations ranging from singleorgan involvement to generalised disease. Given the rarity of the disease, most of the information regarding it comes from case reports. To date, no clinical trials concerning treatment are available. This review focuses on thoracic GLA and summarises possible diagnostic and therapeutic approaches. @ERSpublications Possible diagnostic and therapeutic approaches to generalised lymphatic anomaly (lymphangiomatosis)

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“…Lymphangiomatosis is also associated with chylopericardium, 13 chyloptysis, 14 hemoptysis, 15 chylous ascites, [16][17][18] and peripheral lymphedema. [19][20][21] Purulent pleural fluid (empyema) can also appear white. Therefore pleural fluid triglyceride levels should be obtained for confirmation (a level greater than 110 mg/dL is highly suggestive of chylothorax, whereas levels below 50 mg/dL virtually exclude the diagnosis).…”

Section: Pleural Effusionmentioning

confidence: 99%

Thoracic Lymphatic Disorders

Davis

Berry²,

Raffin³

et al. 2004

Lymphatic Research and Biology

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Thoracic complications of lymphatic disorders can culminate in respiratory failure and death and should be considered in any patient with a lymphatic disease and clinical or radiographic evidence of chest disease. Congenital lymphatic disorders are being increasingly recognized in the adult population. The spectrum of thoracic manifestations of lymphatic disorders ranges from incidental radiographic findings to diffuse lymphatic disease with respiratory failure. This article serves to review some recent advances that allow improved diagnosis and management of thoracic lymphatic disorders. Herein, we describe their anatomical and physiologic effects, the time course of their progression, and the therapies that are currently available. The management of malignant (cancerous) lymphatic disorders of the thorax is beyond the scope of this paper.

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Chylous Pleural Effusion Associated With Primary Lymphedema and Lymphangioma-like Malformations

Cited by 10 publications

References 14 publications

Pseudochylothorax in pleural effusion due to coronary artery bypass surgery

Pseudochylothorax in pleural effusion due to coronary artery bypass surgery

Thoracic involvement in generalised lymphatic anomaly (or lymphangiomatosis)

Thoracic Lymphatic Disorders

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