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Ann Thorac Surg 1998;65:1461-1464
© 1998 The Society of Thoracic Surgeons

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Case Reports

Giant Solitary Fibrous Tumor of the Pleura

^a Department of Thoracic Surgery, Mount Zion-UCSF Cancer Center, San Francisco, California, USA
^b Department of Medicine, Mount Zion-UCSF Cancer Center, San Francisco, California, USA
^c Department of Interventional Radiology, Mount Zion-UCSF Cancer Center, San Francisco, California, USA
^d Department of Pathology, Mount Zion-UCSF Cancer Center, San Francisco, California, USA

Accepted for publication December 14, 1997.

	Abstract

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Solitary fibrous tumors of the pleura are rare. Approximately 600 cases have been described in the literature. We report a case of a young man with a giant solitary fibrous tumor of the pleura that filled his entire left hemithorax and anterior mediastinum and extended into the right side of his chest. The diagnostic modalities employed, the operation, and the postoperative management resulting in complete resection of the tumor and full lung reexpansion are described.

	Introduction

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A 44-year-old man presented with progressively increasing shortness of breath. A chest radiograph and computed tomographic scan revealed a large, heterogeneous mass in the left chest displacing the mediastinal structures to the right, involving the lung parenchyma and hilum with some central calcifications. After a negative tuberculosis skin test, results of a fine-needle aspiration of the mass led us to suspect benign fibrous tumor of the pleura.

Past medical history was significant for 40 pack-years of tobacco and alcohol use. Physical examination revealed a well-nourished, thin man in no acute distress. Significant findings included absence of breath sounds in the left hemithorax and hepatosplenomegaly. Laboratory analysis included the following results: arterial oxygen tension, 78 mm Hg; arterial carbon dioxide tension, 40 mm Hg; prothrombin time, 15.1 seconds; platelet count, 98 x 10³/mL; glucose level, 100 mg/dL; forced expiratory volume in 1 second, 1.1 L (69%): and diffusing capacity for carbon monoxide, 90%. Hematologic evaluation led us to conclude that splenomegaly was the cause of the thrombocytopenia and no other preoperative measures were indicated.

Bronchoscopy revealed a rightward deviation of the trachea and extrinsic compression causing obstruction of the distal left main bronchus. A extended posterolateral thoracotomy was initially performed. The tumor was successfully mobilized posteriorly and inferiorly off the lower lobe and diaphragm. However, the tumor was firmly adherent in the apex and centrally so that proximal control at the hilum was not possible because of the large size of the tumor. We elected to close the thoracotomy, leaving the tumor in place. After the patient had fully recovered, magnetic resonance imaging was performed to better assess mediastinal and apical involvement (Fig 1) in planning resection from an anterior approach. Compression of mediastinal structures was noted, and aortic invasion could not be excluded. An aortogram did not demonstrate aortic invasion and also delineated multiple feeding vessels arising from the left subclavian artery and thyrocervical trunk to the tumor, which were successfully embolized by interventional radiology (Fig 2). A balloon catheter was inserted through the right femoral artery and positioned in the subclavian artery before the vertebral artery take-off for intraoperative proximal control.

View larger version (148K): [in this window] [in a new window]   Fig 1. Preoperative magnetic resonance image shows possible aortic invasion.

View larger version (79K): [in this window] [in a new window]   Fig 2. Aortogram and subsequent embolization of tumor feeding vessels.

The tumor measured 26.0 x 24.0 x 12.0 cm. A partial parietal pleurectomy was performed to minimize the chances of local recurrence. Pathologic examination demonstrated a solitary, well-circumscribed, lobulated mass. On histologic examination the tumor exhibited a classic "patternless" pattern of a solitary fibrous tumor of the pleura with an interweaving network of fibroblastlike cells (Fig 3), accompanied by the deposition of abundant collagen. The margins were negative for tumor.

View larger version (131K): [in this window] [in a new window]   Fig 3. Pathologic specimen shows an interwoven network of fibroblastlike cells, abundant collagen, and a lack of nuclear abnormality and mitotic figures. (Hematoxylin and eosin; x100 before 54% reduction.)

View larger version (165K): [in this window] [in a new window]   Fig 4. Chest radiograph 3 weeks postoperatively shows good reexpansion of the right lung.

	Comment

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The usual presentation is an asymptomatic mass discovered incidentally on a chest radiograph [2]. However, cough, chest pain, dyspnea, and clubbing are seen in 30% of the patients and are more common with tumors greater than 10 cm and those with malignant cellular features. Hypoglycemia has also been noted in some very large tumors because of release of insulinlike peptide. Cytologic examination of fine-need aspirate, sputum, and pleural fluid usually is not helpful in making a diagnosis. Final diagnosis requires surgical resection to determine the presence of malignant components.

At this time there is no clear agreement as to the true histologic differences in the origin of localized and diffuse mesothelioma. The term "benign localized tumor of the pleura" has been proposed by some to recognize the clearly different pathogensis between localized and diffuse mesothelioma. Grossly, the tumors are encapsulated, firm, lobulated masses with a characteristic whorled appearance in the benign tumor and a more homogeneous texture in the malignant form. Areas of calcifications, hemorrhage, and necrosis also may be seen. These tumors are usually less than 10 cm in size and are pedunculated. Eighty percent originate from the visceral pleura and 20% from the parietal pleura [1, 2]. Although most solitary fibrous tumors are benign, up to 13% of the localized tumors can have malignant characteristics and a more aggressively fatal course [2]. The diagnosis of malignancy is difficult without direct tissue invasion but usually is based on cellular atypia and mitotic rate. The localized tumors are more common in women (2:1) versus the preponderance of diffuse mesothelioma in men, which parallels exposure risks. There is no link to tobacco or asbestos in the localized tumor, whereas the link between asbestos and diffuse mesothelioma has been clearly established [1].

Chest radiography and computed tomographic scanning are the imaging studies of choice. Magnetic resonance imaging is helpful for assessing possible vascular invasion, although angiograms are more definitive. Occasionally angiography is helpful to delineate major feeding vessels, as demonstrated here, and it can be used for possible embolization of very large tumors.

Optimal treatment of benign localized pleural tumors entails complete excision. The overall operative mortality is reported at 12% because of hemodynamic changes associated with decompressing the mediastinal structures [2]. Localized chest wall invasion does not necessarily equate with malignant tumor, and the attached chest wall should be resected where necessary with the tumor, especially in tumors arising from the parietal pleura. The underlying lung parenchyma usually can be preserved, but clear margins must be obtained. If, however, the tumor arises from the visceral pleura, adequate en bloc wedge resection of underlying lung should be done.

Local recurrence even after complete resection has been reported as late as 17 years postoperatively [6]. Re-resection is indicated in the absence of disseminated disease and has not adversely affected survival in patients with locally recurrent benign tumors [1, 6].

Survival is directly related to whether the tumor can be resected completely and the presence of malignant changes histologically. The role of adjuvant therapy has not been determined because of the rarity of the lesion [7]. In most cases, however, because of the low cellular content and low mitotic rate of these tumors, chemotherapy and radiotherapy are not effective.

	References

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1. Antman K.H. Clinical presentation and natural history of benign and malignant mesothelioma. Semin Oncol 1981;8:313-320.[Medline]
2. Briselli M., Mark E.J., Dickersin G.R. Solitary fibrous tumors of the pleura: eight new cases and review of 360 cases in the literature. Cancer 1981;47:2678-2689.[Medline]
3. Martini N., McCormack P.M., Bains M.S., et al. Pleural mesothelioma. Ann Thorac Surg 1987;43:113-120.[Abstract]
4. Watts D.M., Jones G.P., Bowman G.A., Olsen J.D. Giant benign mesothelioma. Ann Thorac Surg 1989;48:590-591.[Abstract]
5. Scharifker D., Kaneko M. Localized fibrous "mesothelioma" of pleura (submesothelial fibroma): a clinicopathologic study of 18 cases. Cancer 1979;43:627-635.[Medline]
6. Okike N., Bernatz P.E., Woolner L.B. Localized mesothelioma of the pleura: benign and malignant variants. J Thorac Cardiovasc Surg 1978;75:363-372.[Abstract]
7. McCormack P.M., Nagasaki F., Hilaris B.S., Martini N. Surgical treatment of pleural mesthelioma. J Thorac Cardiovasc Surg 1982;84:834-842.[Abstract]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Junaid H. Khan Timothy S. Hall David M. Jablons Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Khan, J. H. Articles by Jablons, D. M. Search for Related Content PubMed PubMed Citation Articles by Khan, J. H. Articles by Jablons, D. M.