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Ann Thorac Surg 1999;67:1456-1459
© 1999 The Society of Thoracic Surgeons

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Original Articles

Clinical behavior of solitary fibrous tumors of the pleura

^a Unit of Thoracic Surgery, Department of Surgery, University Hospital of Geneva, Geneva, Switzerland
^b Division of Clinical Pathology, University Hospital of Geneva, Geneva, Switzerland

Accepted for publication November 23, 1998.

Address reprint requests to Dr Spiliopoulos, Thoracic Surgery, Department of Surgery, University Hospital of Geneva, rue Micheli-du-Crest 24, 1211 Geneva 14, Switzerland

	Abstract

Top Abstract Introduction Material and methods Results Discussion References

 
Background. Solitary fibrous tumors of the pleura are rare and present unpredictable clinical behavior.

Methods. Between 1981 and 1998, 11 solitary fibrous tumors of the pleura were resected in 10 patients at the University Hospital of Geneva. Their clinical behavior and outcome were reviewed.

Results. Seven tumors arose from the visceral pleura, and three arose from the parietal pleura. Tumors arising from the parietal pleura were revealed to be more difficult to resect than those from the visceral pleura because of their size and adhesion to the chest wall requiring extrapleural resection. Eight tumors showed benign features, whereas two showed distinct features of malignity. One additional patient presented marked pleomorphism that could represent an intermediate form before frank malignity. Four tumors had been followed expectandly for 2 to 10 years before surgery. Although three enlarged rapidly, no signs of malignity were observed on histological examination. All patients are alive, from 2 months to 14 years after surgery (mean 55 months). In one case, however, a malignant tumor recurred 6 years after resection of a benign variant.

Conclusions. Although histologically benign, solitary fibrous tumors of the pleura may enlarge rapidly and occasionally transform into malignant variants after several years. Therefore, complete surgical resection and long-term follow-up is recommended for all patients.

	Introduction

Top Abstract Introduction Material and methods Results Discussion References

 
Primary neoplasms of the pleura are rare tumors with unpredictable behavior. Benign forms may recur and occasionally become malignant, whereas malignant variants may be cured by simple excision [1, 2]. England and associates [1] reviewed 223 cases and found that resectability was the single most important indicator of good clinical outcome. Surgical exploration is usually carried out shortly after discovery, because malignity cannot be excluded on imagery alone. The purpose of this study was to summarize our experience in the management of solitary fibrous tumors of the pleura and to review their clinical behavior and outcome.

	Material and methods

Top Abstract Introduction Material and methods Results Discussion References

 
Between 1981 and 1998, 11 solitary fibrous tumors of the pleura were resected in 10 patients at the University Hospital of Geneva. The medical records were reviewed, and follow-up was obtained by repeated examination of the patients or contact with the patient or his physician.

The tumors were classified into benign or malignant variants. The benign variant was a firm, usually encapsulated and lobulated tumor with a fibrous whorled appearance and occasional hemorrhage or necrosis on cut surface. Histologically, it was characterized by a proliferation of uniform elongated spindle cells with a various amount of connective tissue. Fibrous or acellular, cellular, and mixed bundles of tissue were seen in different sampled areas of the tumor. Slight to marked amounts of blood vessels were observed (Fig 1). The malignant variant was macroscopically similar to the benign one, but contained high mitotic activity defined as at least four mitotic figures per 10 high-power fields (HPF) and mild to marked pleomorphism based on nuclear size, irregularity, and nucleolar prominence. Bundles of fusocellular cells with high cellularity as well as crowding and overlapping of nuclei ressembling fibrosarcoma could occasionally be seen (Fig 2). All solitary fibrous tumors of the pleura were strongly positive for vimentine and CD34, but remained negative for keratin.

View larger version (133K): [in this window] [in a new window]   Fig 1. Benign solitary fibrous tumor of the pleura. Small ovoid or spindle cells without nuclear atypia in a mixoid loose matrix (H&E x100).

View larger version (169K): [in this window] [in a new window]   Fig 2. Malignant solitary fibrous tumor of the pleura. Highly cellular tumor with spindle cells arranged in a storiform pattern (H&E x100).

	Results

Top Abstract Introduction Material and methods Results Discussion References

View larger version (113K): [in this window] [in a new window]   Fig 3. Computed tomography scan showing a large solitary fibrous tumor of the pleura with heterogenous zones due to hemorrhage and necrosis of the tumor. There is no evidence of chest wall invasion (patient 5).

View larger version (121K): [in this window] [in a new window]   Fig 4. Magnetic resonance imaging showing a large solitary fibrous tumor of the pleura situated on the diaphragm (patient 10).

Macroscopic findings and treatment
Six tumors were located in the left hemithorax and six in the right. At surgery, seven tumors arose from the visceral pleura, five were pedunculated, and two presented a broad base of attachment. Two tumors were located in the interlobar fissure. Resection was performed by wedge resection of the lung. Lobectomy and pneumonectomy were never required. Tumors arising from the parietal pleura were more difficult to resect than those from the visceral pleura because they were large, affixed to the chest wall, and required extrapleural resection. Although all tumors originating from the parietal pleura had a broad base of attachment, chest wall resection was never required. In one case, however, the tumor arose beneath the mediastinal reflection of the parietal pleura and required resection of a portion of the diaphragm and lung. Resection was complete in all patients.

Histopathological features
Eight tumors were benign (Table 3). Two tumors showed distinct features of malignity (Table 4). One was a large and nonpedunculated mass arising from the visceral pleura with signs of malignity localized to some areas of the tumor. The second was a recurrent malignant tumor occurring 6 years after resection of a benign one. One tumor had marked pleomorphism in two distinct areas. Since no mitoses were observed, this tumor was not considered as frankly malignant, but such findings could represent an intermediate form before frank malignity (Table 4).

	Discussion

Top Abstract Introduction Material and methods Results Discussion References

 
The clinical behavior of solitary fibrous tumors of the pleura is not well known [3]. Although most tumors are considered histologically benign, local recurrences and rapid enlargement without signs of invasion or metastasis have been reported [4, 5]. In our experience, 4 patients had been followed expectandly between 2 and 10 years before surgery. Although three enlarged rapidly, no signs of malignancy were observed on histological examination. In one case, the tumor presented a broad base of attachement to the parietal pleura and had large adherences to the lung and diaphragm. No histological signs of malignancy were observed after complete resection. However, 6 years later, the tumor recurred with fibrosarcomatous features. Hence, although histologically benign, solitary fibrous tumors of the pleura may enlarge and transform into malignant tumors.

Malignant forms account for approximately 12% of all fibrous tumors of the pleura. They are rarely pedunculated and may arise from the visceral or parietal pleura [2]. Their size is usually greater than 8 cm, and the tumors often present in atypical locations such as the parietal or mediastinal pleuras, lung fissures, and intraparenchymal sites [6–8]. Histological features usually include increased cellularity, pleomorphism, and more than four mitosis per 10 HPF [1]. The number of mitosis is certainly the best indicator of malignity. However, on rare occasions, a marked but isolated pleomorphism may be seen in tumors greater than 8 cm. Although no other signs of malignity are observed, this finding may represent an intermediate variant before frank malignancy. The mortality among malignant fibrous tumors is high, and most patients die within 10 years of surgery unless the tumor is pedunculated [1, 2].

Since fibrous tumors of the pleura may rapidly enlarge and are potentially malignant, surgical resection is recommended in all cases. Preoperative transthoracic needle biopsy is not necessary, because it does not exclude malignant variants and does not influence the need for surgical resection. Care should be taken to remove the whole mass. Hence, although the tumor may be well circumscribed, large resection of lung parenchyma and surrounding pleura is encouraged, in particular with tumors presenting a broad base of attachement to the pleural surface [9]. Postoperative adjuvant therapy with radiotherapy, chemotherapy, or both has been sporadically used, but its benefit remains unproved. Recurrence after complete resection is possible in benign and malignant variants, therefore, long-term annual follow-up with chest roentgenograms is highly recommended.

	References

Top Abstract Introduction Material and methods Results Discussion References

 

1. England D.M., Hochholzer L., McCarthy M.J. Localized benign and malignant fibrous tumors of the pleura. Am J Surg Pathol 1989;13:640-658.[Medline]
2. Okike N., Bernatz P.E., Woolner L.B. Localized mesothelioma of the pleura. Benign and malignant variants. J Thorac Cardiovasc Surg 1978;75:363-370.[Abstract]
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4. Weymand B., Noël H., Goncette L., Noirhomme P., Collard P. Solitary fibrous tumor of the pleura. A report of five cases diagnosed by transthoracic cutting needle biopsy. Chest 1997;112:424-428.
5. Utley J.R., Parker J.C., Hahn R.S., Bryant L.R., Mobin-Uddin K. Recurrent benign fibrous mesothelioma of the pleura. J Thorac Cardiovasc Surg 1973;65:830-834.[Medline]
6. 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]
7. Sanguinetti C.M., Marchesani F., Ranaldi R., Pela R., Cecarini L. Localized fibrous pleural tumour of the interlobular pleura. Eur Respir J 1996;9:1094-1096.[Abstract]
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9. Martini N., McCormack P.M., Bains M.S., Kaiser L.R., Burt M.E., Hilaris B.S. Pleural mesothelioma. Ann Thorac Surg 1987;43:113-120.[Abstract]

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