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Mediastinal lymphangioma and chylothorax: thoracic involvement of Gorham's disease.

Yoo SY, Goo JM, Im JG - Korean J Radiol (2002 Apr-Jun)

Bottom Line: We report a case of mediastinal lymphangioma associated with Gorham's disease in a 38-year-old man who had suffered recurrent clavicular fractures during a seven-year period.Mediastinal widening associated with osteolysis of the clavicles and the sternal manubrium was revealed by chest radiography, while computed tomography demonstrated a cystic anterior mediastinal mass infiltrating mediastinal fat and associated with osseous destruction of the clavicles and manubrium.Chylothorax recurred during the course of the disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Radiology, Seoul National University College of Medicine, Institute of Radiation Medicine, SNUMRC, Clinical Research Institute, Seoul National University Hospital, Seoul, Korea.

ABSTRACT
We report a case of mediastinal lymphangioma associated with Gorham's disease in a 38-year-old man who had suffered recurrent clavicular fractures during a seven-year period. Mediastinal widening associated with osteolysis of the clavicles and the sternal manubrium was revealed by chest radiography, while computed tomography demonstrated a cystic anterior mediastinal mass infiltrating mediastinal fat and associated with osseous destruction of the clavicles and manubrium. Chylothorax recurred during the course of the disease.

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Gorham's disease in a 38-year-old man with mediastinal lymphangioma.A. Posteroanterior chest radiograph shows widening of the mediastinum and osteolysis of the proximal clavicles.B, C. Contrast-enhanced CT scan demonstrates an anterior mediastinal mass of low attenuation, which infiltrates mediastinal fat and envelops adjacent structures. Lysis of the manubrium (arrow in B) is apparent and follow-up CT (C) after three months shows left pleural effusion.D. Irregular-shaped dilated lymphatic spaces lined by attenuated endothelium are embedded in connective tissue stroma, and small lymphoid aggregates are dispersed in the stroma. These findings are compatible with cavernous lymphangioma (original magnification, ×250; hematoxylin-eosin staining).
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Figure 1: Gorham's disease in a 38-year-old man with mediastinal lymphangioma.A. Posteroanterior chest radiograph shows widening of the mediastinum and osteolysis of the proximal clavicles.B, C. Contrast-enhanced CT scan demonstrates an anterior mediastinal mass of low attenuation, which infiltrates mediastinal fat and envelops adjacent structures. Lysis of the manubrium (arrow in B) is apparent and follow-up CT (C) after three months shows left pleural effusion.D. Irregular-shaped dilated lymphatic spaces lined by attenuated endothelium are embedded in connective tissue stroma, and small lymphoid aggregates are dispersed in the stroma. These findings are compatible with cavernous lymphangioma (original magnification, ×250; hematoxylin-eosin staining).

Mentions: Posteroanterior chest radiographs obtained at admission showed bilateral widening of the mediastinum and resorption of the proximal clavicles and the sternal manubrium (Fig. 1A). Postcontrast CT scans demonstrated a well-defined mediastinal mass of low attenuation, infiltrating mediastinal fat and enveloping mediastinal structures. Osteolysis of the clavicles and manubrium was noted (Fig. 1B). The lesion extended superiorly to the internal jugular chain, anteriorly to the pectoralis major and posteriorly to the paratracheal area, without displacing or compressing mediastinal structures. Lymph node enlargement was indefinite. Percutaneous fine needle aspiration of the lesion yielded 340 cc of wine-colored serous fluid: analysis of the aspirate revealed the presence of many lymphocytes and macrophages. There was no evidence of malignancy, and neither special staining nor subsequent culture indicated that microorganisms, including acid-fast bacilli and fungi, were present.


Mediastinal lymphangioma and chylothorax: thoracic involvement of Gorham's disease.

Yoo SY, Goo JM, Im JG - Korean J Radiol (2002 Apr-Jun)

Gorham's disease in a 38-year-old man with mediastinal lymphangioma.A. Posteroanterior chest radiograph shows widening of the mediastinum and osteolysis of the proximal clavicles.B, C. Contrast-enhanced CT scan demonstrates an anterior mediastinal mass of low attenuation, which infiltrates mediastinal fat and envelops adjacent structures. Lysis of the manubrium (arrow in B) is apparent and follow-up CT (C) after three months shows left pleural effusion.D. Irregular-shaped dilated lymphatic spaces lined by attenuated endothelium are embedded in connective tissue stroma, and small lymphoid aggregates are dispersed in the stroma. These findings are compatible with cavernous lymphangioma (original magnification, ×250; hematoxylin-eosin staining).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC2713836&req=5

Figure 1: Gorham's disease in a 38-year-old man with mediastinal lymphangioma.A. Posteroanterior chest radiograph shows widening of the mediastinum and osteolysis of the proximal clavicles.B, C. Contrast-enhanced CT scan demonstrates an anterior mediastinal mass of low attenuation, which infiltrates mediastinal fat and envelops adjacent structures. Lysis of the manubrium (arrow in B) is apparent and follow-up CT (C) after three months shows left pleural effusion.D. Irregular-shaped dilated lymphatic spaces lined by attenuated endothelium are embedded in connective tissue stroma, and small lymphoid aggregates are dispersed in the stroma. These findings are compatible with cavernous lymphangioma (original magnification, ×250; hematoxylin-eosin staining).
Mentions: Posteroanterior chest radiographs obtained at admission showed bilateral widening of the mediastinum and resorption of the proximal clavicles and the sternal manubrium (Fig. 1A). Postcontrast CT scans demonstrated a well-defined mediastinal mass of low attenuation, infiltrating mediastinal fat and enveloping mediastinal structures. Osteolysis of the clavicles and manubrium was noted (Fig. 1B). The lesion extended superiorly to the internal jugular chain, anteriorly to the pectoralis major and posteriorly to the paratracheal area, without displacing or compressing mediastinal structures. Lymph node enlargement was indefinite. Percutaneous fine needle aspiration of the lesion yielded 340 cc of wine-colored serous fluid: analysis of the aspirate revealed the presence of many lymphocytes and macrophages. There was no evidence of malignancy, and neither special staining nor subsequent culture indicated that microorganisms, including acid-fast bacilli and fungi, were present.

Bottom Line: We report a case of mediastinal lymphangioma associated with Gorham's disease in a 38-year-old man who had suffered recurrent clavicular fractures during a seven-year period.Mediastinal widening associated with osteolysis of the clavicles and the sternal manubrium was revealed by chest radiography, while computed tomography demonstrated a cystic anterior mediastinal mass infiltrating mediastinal fat and associated with osseous destruction of the clavicles and manubrium.Chylothorax recurred during the course of the disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Radiology, Seoul National University College of Medicine, Institute of Radiation Medicine, SNUMRC, Clinical Research Institute, Seoul National University Hospital, Seoul, Korea.

ABSTRACT
We report a case of mediastinal lymphangioma associated with Gorham's disease in a 38-year-old man who had suffered recurrent clavicular fractures during a seven-year period. Mediastinal widening associated with osteolysis of the clavicles and the sternal manubrium was revealed by chest radiography, while computed tomography demonstrated a cystic anterior mediastinal mass infiltrating mediastinal fat and associated with osseous destruction of the clavicles and manubrium. Chylothorax recurred during the course of the disease.

Show MeSH
Related in: MedlinePlus