Chylothorax complicating Gorham’s disease

Chavanis, Nicolas; Chaffanjon, Philippe; Frey, Gil; Vottero, G; Brichon, Pierre-Yves

doi:10.1016/s0003-4975(00)02417-6

Cited by 35 publications

(32 citation statements)

References 5 publications

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“…However, the Gorham's disease should be included in differential diagnosis of chylothorax, especially when it is associated with osteolytic lesion. There have been about 30 cases of chylothorax complicating Gorham's disease in the English literature (4,8,9).…”

Section: Discussionmentioning

confidence: 99%

“…For the prompt control of chylothorax, the available evidence suggests that early surgical intervention to ligate the thoracic duct reduce mortality related with malnutrition and sepsis, even though there are only a small number of cases reported (4,9,10). Ligation of thoracic duct and excision of the pathologic lymphagiomatous tissue is indicated (4).…”

Section: Discussionmentioning

confidence: 99%

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Chylothorax in Gorham's Disease

Lee

Kim

et al. 2002

J Korean Med Sci

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A 25-yr-old woman presented with a right pleural effusion. Destruction of 9th through 12th ribs, adjacent vertebral bodies, and transverse processes was noted on plain radiograph and a large low-attenuated, irregular shaped mass lesion with peripheral rim enhancement, destroying vertebral body and transverse process, was revealed on the computed tomographic scan. Magnetic resonance imaging showed high signal on T1- weighted image and iso- and low signal on T2-weighted image for the mass lesion replacing the vertebral bony cortex and marrow space. An open rib biopsy revealed the histopathological changes of Gorham's disease (essential osteolysis), even though only bloody fluid filling the empty space and rib and vertebral transverse process destruction were grossly observed on operation. Even though there was no definite response to radiotherapy and pleurodesis, the patient showed stable condition up to 20 months after diagnosis.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Chylothorax in Gorham's Disease

Lee

Kim

et al. 2002

J Korean Med Sci

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“…Others have regarded chylothorax to be related to disruption to the thoracic duct or pleural lymphatic vessels by adjacent osteolysis. 2,21 In the presented case, continuous progression of the disease could be observed despite administration of experimental medical therapy. After vertebral body fractures and transpedicular stabilization, the patient was treated with IFN-α2b and bisphosphonates.…”

Section: Discussionmentioning

confidence: 71%

Cerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine

Molina¹,

Niederstadt²,

Ruland

et al. 2014

SPI

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Patients with Gorham-Stout disease (GSD), a rare disease of poorly understood etiopathophysiology, suffer from progressive osteolysis. Destruction of bone matrix is caused by lymphatic vessels, which can lead to CSF leakage if parts of bony structures adjacent to CSF spaces are involved. So far, fewer than 200 patients have been reported in the literature; only 4 of these patients presented with CSF leakage. The authors report the case of a 30-year-old man with GSD and CSF leakage due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine. Neurosurgical intervention, including dura repair, was needed. Experimental medical therapy with rapamycin was started, leading to disease control for more than 12 months. Progression of GSD can lead to destruction of the meninges, causing CSF leakage. The authors review 4 other cases reported in the literature and discuss therapeutic options.

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“…Moreover in other cases, a proliferation of lymphatic vessels was demonstrated in histological specimens of the pleura [15-18, 22, 26, 29, 31, 39]. Sometimes the bone lesions are localized remote from the chest [7,16,21,39] in these cases the pathogenetic mechanisms of chylothorax is completely unknown and an undefined systemic process damaging the thoracic duct or other lymphatic vessels can be only supposed. Further studies will be needed to elucidate this aspect.…”

Section: Discussion and Literature Reviewmentioning

confidence: 99%

Pleural Effusion in Gorham-Stout Syndrome: A Case Report and a Literature Review

2015

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Gorham-Stout syndrome (GSS) is an uncommon disease characterized by massive osteolysis due to bone resorption associated with proliferation of blood or lymphatic vessels. Pleural effusion is an uncommon manifestation. We present a case report of GSS in a 46Keywords: Pleural effusion, Gorham-stout syndrome, Pleural disease, Chylothorax, Osteolysis, Treatment strategy. 2015 Vol.2, No 2, pp.11-22 ISSN(e): 2408-9273 ISSN(p): 2409-2053 DOI: 10.18488/journal.95/2015.2.2/95.2.11.22 © 2015 Cancers Review Contribution/ OriginalityThis study contributes to the existing literature by presenting the first case of bilateral serohematic pleural effusion in Gorham-Stout syndrome that was investigated with chest ultrasound and medical thoracoscopy bilaterally. Furthermore, this study provides the first review of clinical cases reporting pleural effusion secondary to Gorham-Stout syndrome.

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Chylothorax complicating Gorham’s disease

Cited by 35 publications

References 5 publications

Chylothorax in Gorham's Disease

Chylothorax in Gorham's Disease

Cerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine

Pleural Effusion in Gorham-Stout Syndrome: A Case Report and a Literature Review

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