Ann Thorac Surg 2003;76:892-895
© 2003 The Society of Thoracic Surgeons
Original article: general thoracic
Localized fibrous tumors of the pleura: clinical and surgical evaluation
Tamer Altinok, MDa*,
Salih Topçu, MDb,
A. Irfan Tastepe, MDa,
Ulkü Yazici, MDa,
Güven Çetin, MDa
a Atatürk Center for Chest Disease and Thoracic Surgery, Department of Thoracic Surgery, Keçiören-Ankara, Turkey
b Kocaeli University, School of Medicine, Department of Thoracic Surgery, Kocaeli, Turkey
Accepted for publication March 25, 2003.
* Address reprint requests to Dr Altinok, 
ehit Mustafa Ba
Cad. 79/4, Aydinlikevler 06130, Ankara, Turkey.
e-mail: taltinoks{at}yahoo.com
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Abstract
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BACKGROUND: Localized fibrous tumors of the pleura, which are considered to originate from submesothelial connective tissue, are rare. The purpose of this study was to investigate the clinical features of these tumors and to determine the optimal treatment and follow-up.
METHODS: The records of 10 consecutive patients with localized fibrous tumors of the pleura operated on at the Ataturk Center for Chest Diseases and Thoracic Surgery between 1995 and 2001 were retrospectively reviewed. Diagnostic procedures, clinical courses, and outcomes of these patients were studied. Total excision through a thoracotomy was performed in all of the patients. Pneumonectomy was required in 1 patient because of a giant intraparenchymal localized fibrous tumor of the pleura. Additional partial chest wall resections were done in 2 patients.
RESULTS: The size of excised tumors ranged from 10 x 8 x 4 cm to 30 x 25 x 20 cm. Malignant transformation was seen in 1 patient 22 months after resection of a benign tumor. Four cases were pathologically considered to be malignant. All patients except 1 are alive (follow-up 16 to 57 months after resection).
CONCLUSIONS: Although localized fibrous tumors of the pleura are considered histologically benign tumors, because of the risk of recurrence and malignant transformation, complete surgical resection is indicated and long-term follow-up is recommended in all patients.
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Introduction
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Localized fibrous tumors of the pleura (LFTPs) are rare tumors that are considered to develop from submesothelial connective tissue [1, 2]. The LFTPs can usually be distinguished from malignant mesothelioma by their radiographic features, gross appearance (often pedinculated), immunohistologic characteristics, and ultrastructural characteristics. Thirteen percent of reported tumors had aggressive clinical behavior with local infiltration and local recurrence. The remaining 87% had benign clinical behavior and were resected completely, including adjacent structures if necessary, which is generally believed to be sufficient treatment [3, 4].
Ten cases with LFTPs operated in our clinic are reported here because of their different clinical behaviors.
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Material and methods
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Between 1995 and 2001, 10 patients with LFTPs (3 men, 7 women) underwent 11 operations at our clinic. The mean age was 64.1 years (range, 25 to 71 years).
Chest pain (in 10 patients), dyspnea (in 8 patients), cough (in 4 patients), and weight loss (in 4 patients) were the symptoms. On physical examination, dullness to percussion and absence of breath sounds in the ipsilateral hemithorax were detected in all patients, and clubbing was present in 2 patients (Table 1). Chest roentgenogram and computed tomography revealed well circumscribed, heterogeneous, hypodense soft tissue masses in most patients. Pleural effusion was detected by chest roentgenogram in 2 patients. To evaluate the invasion to the chest wall and adjacent structures, no additional radiographic examination, such as magnetic resonance imaging, was required. Fiberoptic bronchoscopy was performed in 5 patients. In all of them, extrinsic compression of segmental bronchi was observed, but no endobronchial lesion was detected.
Thoracocentesis and transparietal biopsy were done in 2 patients with pleural effusion. Sanguinous effusion (2200 mL) and exudative pleural effusion (800 mL) were evacuated in those patients. Because cytologic examination was considered as suspicious for malignancy and transparietal biopsy revealed benign pleuritis in the first patient, diagnostic video-assisted thoracoscopy was performed. The frozen section examination revealed that the mass was malignant. In the second patient with pleural effusion, two distinct masses, 15 x 10 x 10 cm and 12 x 8 x 4 cm, originating from the visceral pleura, were detected.
Transparietal biopsy was performed in 7 patients. In 1 patient, pathologic examination revealed mesenchymal tumor of dubious nature, whereas fibrocollagenous tissue was present in the others.
Five left and five right posterolateral thoracotomies were performed in 10 patients. Reoperation was needed in 1 patient after 22 months because of tumor recurrence. In 7 patients, pedunculated tumors arising from visceral pleura were detected. Total excision was performed easily in those patients because of mild fibrous adhesions. Only 1 patient, who had a giant intraparenchymal mass in the left upper lobe, underwent a pneumonectomy. In 2 patients additional en bloc chest wall resection was required because of suspicion of invasion to the parietal pleura and chest wall. Two ribs were resected in both of them (Fig 1).
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Fig 1. Chest roentgenogram (top left). Computed tomogram revealing a broad-based mass on the parietal pleura and chest wall (top right). En bloc resection was done (bottom left). Gross morphologic image of the mass and ribs (bottom right).
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Results
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The largest mass excised in our patients was 30 x 25 x 20 cm and 4,750 g (Fig 2).
The smallest one was 10 x 8 x 4 cm. Four patients (40%) had malignant and 6 patients (60%) had benign LFTP. Postoperative radiotherapy was used in only 1 patient who had a recurrence.
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Fig 2. Chest roentgenogram (top left). Computed tomogram showing giant mass nearly filled the right hemithorax (top right). The specimen was 4750 g and 30 x 25 x 20 cm (bottom).
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Tumor recurrence was detected in the patient who had two distinct masses and for whom total excision was performed in 22nd month after the first operation. The recurrent tumor (15 x 8 x 6 cm) was removed by rethoracotomy. Histopathologic examination revealed malignant pleural fibroma. Unfortunately, in the 38th month of follow-up, she was admitted to the hospital because of an extraparenchymal giant lobulated mass in the same hemithorax, but she refused reoperation.
One patient died on the 12th postoperative day of empyema and sepsis. The others are alive and do not show any complication or recurrence in the mean 34.4 months (16 to 57 months) of follow-up except the patient with a second recurrence.
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Comment
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Localized fibrous tumors of the pleura are uncommon, representing less than 5% of all neoplasms involving the pleura [3, 5]. Although it is believed that a history of asbestos exposure is lacking in patients with LFTP, 2 of our patients with malignant tumor had history of asbestos exposure.
Localized fibrous tumors of the pleura are frequently asymptomatic and discovered on routine chest radiographs. Extrathoracic manifestations can include arthritic pain, digital clubbing, hypoglycemia, and galactorrhea [2, 4, 6, 7]. We detected clubbing in 2 patients, which regressed completely after surgical resection.
Radiologically, LFTPs appear as solitary, sharply defined or lobulated masses at the periphery of the lung with no evidence of invasion, with or without pleural effusion. Nevertheless, we observed an intraparenchymal mass which was very close to the main vascular structures and required pneumonectomy for complete resection.
Macroscopically, tumors are round or ovoid, encapsulated or sharply circumscribed and attached to visceral pleura often by a pedicle. The size of the tumors has been reported to be from 1 to 39 cm, and the average weight 100 to 400 g, but tumors greater than 10 cm are more likely to be malignant [8]. In our series, all patients had masses greater than 10 cm. The largest mass in our series was 30 x 25 x 20 cm and 4,750 g. Although it was the heaviest mass reported in the literature, it was histologically benign.
Although a report of two cases in which the diagnosis was obtained by fine-needle aspiration biopsy has been published [5], in our series only one transparietal biopsy was found to be a mesenchymal tumor of dubious nature; in the other six it was nondiagnostic. Because of acellular and hypercellular parts of the tumor, it is usually difficult to obtain sufficient material for cytologic analysis [9]. Nevertheless, Weynand and colleagues [10] reported that transthoracic core biopsies with cutting needles were often definitive for diagnosis of LFTPs.
Malignant tumors comprise about 20% of all LFTPs. Sometimes those can be called pleural fibrosarcoma or malignant pleural fibroma [8]. Four tumors in our patients were considered malignant because of features that suggested malignancy, eg, large size (broad based on the parietal pleura), the presence of abnormal mitotic figures, and areas of necrosis. The remaining tumors were considered benign because of the slow rate of growth and the lack of any other features of malignancy except large size and necrosis. It was reported that necrosis can occur because of large size rather than intrinsic malignant potential. Pleural effusion can be seen in both benign and malignant forms, but it usually indicates a greater likelihood of malignancy [8]. We detected sanguinous effusion and serous exudate effusion in 2 patients with malignant tumors.
Multiple tumors are extremely rare [11, 12]. In the series of 55 cases reported by Cardillo and coworkers [13], three synchronous fibrous tumors from the visceral pleura of the same lobe were observed in 1 patient (1.81%). In our series, 1 patient had two synchronous pedunculated fibrous tumors arising from visceral pleura of different lobes. Despite total excision, recurrence and malignant transformation were observed.
The treatment for these tumors is surgical excision. It is easy to perform total excision in most benign lesions. More radical approaches are needed for malignant tumors, for tumors with a broad-based attachment to pleural surfaces, or for intraparenchymal lesions. Approximately 10% of the benign LFTPs recur with malignant change rarely supervening [4]. It was reported that 8 patients with a malignant tumor had recurrence or distant metastasis between 6 months and 8 years after initial resection. The best predictor of a benign course is complete excision with microscopically free surgical margins [11, 14]. We have observed no recurrence in 2 patients with malignant LFTP who underwent en bloc additional chest wall resection. No distant metastasis was detected in any patient.
The prognosis depends on the resectability and size of the tumor, the number of mitoses, polymorphism, and necrosis in the tumor [2]. Postoperative adjuvant radiotherapy, chemotherapy, or both have been used sporadically. Isolated reports have recommended the use of postoperative radiotherapy. However there are no clear data confirming its benefit [11, 14]. Radiotherapy was required in the patient with a second recurrence after reoperation, but we were not able to determine the benefit of radiotherapy.
In conclusion, complete resection and close follow-up for years after operation is recommended for LFTPs. Surgical resection should be considered for recurrence or malignant transformation of these tumors.
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Acknowledgments
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We sincerely thank Dr . Tuba Liman for her skillful preparation of the manuscript.
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References
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Abstract
Introduction
