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Ann Thorac Surg 2005;79:303-307
© 2005 The Society of Thoracic Surgeons

Original article: General thoracic

Solitary Fibrous Tumors of the Pleura: Surgical Outcome and Clinical Course

Sook Hwan Sung, MDa, Jee-Won Chang, MDb, Jhingook Kim, MDb, Kyung Soo Lee, MDc, Jungho Han, MDd, Seung Il Park, MDe,*

a Department of Thoracic Surgery, Seoul National University
b Department of Thoracic Surgery
c Department of Radiology
d Department of Pathology, Samsung Medical Center, Sungkyunkwan University
e Department of Thoracic and Cardiovascular Surgery, Asan Medical Center, University of Ulsan, Seoul, South Korea

Accepted for publication July 6, 2004.

* Address reprint requests to Dr Park, Department of Thoracic and Cardiovascular Surgery, Asan Medical Center, University of Ulsan, 388-1 Poongnap-dong, Songpa-gu, Seoul 138-736, South Korea (E-mail: sipark{at}amc.seoul.kr).


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: The aim of this study was to define more precisely the surgical outcome and clinical course of solitary fibrous tumors of the pleura.

METHODS: We conducted a retrospective review of the clinical records of patients who had undergone surgical resection for benign and malignant solitary fibrous tumors of the pleura during a 10-year period (1993 to 2003).

RESULTS: Sixty-three patients were enrolled in the study (men, 29; women, 34; mean age, 49.6 years). Thirty-six patients (57.1%) were symptomatic at the time of diagnosis. Resection was performed through a thoracotomy (n = 37), by means of video-assisted thoracoscopy (n = 22), or through a sternotomy (n = 4). Mass excision only was performed in 34 cases, and en bloc excision including adjacent structures was performed in 29 cases. Forty-four cases (69.8%) were benign and 19 (30.2%) were malignant. Local recurrences occurred in three cases and distant metastases in eight. Recurrences occurred only in malignancy. Symptomatic presentation and the impression of a nonpleural tumor by imaging study were found to be related to a malignant pathologic diagnosis. The radiologic impression of solitary fibrous tumors of the pleura was also related to mass excision only.

CONCLUSIONS: For malignant cases, complete surgical resection may be insufficient for the cure. Therefore, further study should be performed to define the role of preoperative and postoperative systemic treatment.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 References
 
Solitary fibrous tumors of the pleura (SFTP) are rare neoplasms that usually originate from the visceral pleura. Since its pathologic characteristics was first described by Klemperer and Rabin in 1931 [1], the nomenclature has become confused, and the disease has also been referred to as a localized mesothelioma, localized fibrous tumor, fibrous mesothelioma, or a pleural fibroma [2]. The further development of electron microscopy and immunohistochemistry has clarified that the tumor does not originate from the mesothelial layer but from the submesothelial, noncommitted mesenchymal layer [3, 4]. Thus, the various names used for this disease have become unified, and the disease is now referred to as solitary or localized fibrous tumors of the pleura. Mesenchyme is pluripotent tissue and possesses diverse differentiation potential to bone, cartilage, or blood vessels. Because of this diversity of the mesenchyme, the pathologic morphology of SFTP appears variable [5]. Recent studies on SFTP have mainly involved immunologic markers for pathologic diagnosis [6, 7] and some clinical reports that include postsurgical resection results and description of its clinical behavior [8–10].

Although complete surgical resection of benign SFTP is the usual method of cure, occasional reports advise caution concerning its unpredictable clinical behavior such as its invasion of adjacent organs [11] or cardiac compression by the huge mass of benign SFTP [12]. Furthermore, the number of reported cases of malignant SFTP is too small to offer an overview, recurrence or survival are inconsistently reported, and no established treatment modality or follow-up plan has been agreed to.

The aim of this study was to assess more precisely clinical behavior, surgical outcome, and the propriety of surgical techniques. This study also includes the determination of whether clinical or radiologic information can predict postoperative results.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 References
 
We retrospectively reviewed the clinical records of patients who had undergone surgical resection for benign or malignant SFTP from 1993 to 2003. We reexamined pathologic slides, and malignant SFTP was diagnosed when one of following criteria was satisfied [10]; (1) increasing mitotic activity (>4 mitoses per 10 high-power fields), (2) high cellularity with crowding and overlapping of nuclei, (3) presence of necrosis, or (4) pleomorphism.

History taking, a physical examination, a routine blood test, standard chest radiography, electrocardiography, and a thoracic computed tomographic scan were available for all patients. Lung perfusion scans or echocardiographic results were available in selected cases.

Recent patient status was determined by using the clinical records of outpatient clinics or by telephone interview. Operative mortality was defined as death within 30 days of an operation or during hospitalization.

The continuity-corrected {chi}2 test was used to compare percentages. Survival was analyzed with Kaplan-Meier survivor function estimates. A p value of less than 0.05 was considered statistically significant.


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 References
 
Sixty-three patients underwent surgical resection for a benign or malignant SFTP during the 10-year period. There were 29 men (46%) and 34 women (54%) of a mean age of 49.6 years (range, 5 to 83 years). No patients had a history of asbestos exposure.

Thirty-six patients (57.1%) were symptomatic at the time of diagnosis, and the most common symptom was dyspnea. One patient presented with hypertrophic osteoarthropathy, but no patient presented with symptomatic hypoglycemia (Table 1). The remainder of the patients were absolutely asymptomatic; tumors were incidentally found during standard chest radiography.


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  		Table 1. Patient Characteristics
 
The most favorable impression of the radiologist based on a thoracic computed tomographic scan was SFTP or a pleural mass in 36 patients (57%), and the remainder were impressions of a pulmonary mass (lung cancer, sclerosing hemangioma, angiolipoma) or mediastinal tumor (thymoma, neurogenic tumor, submucosal esophageal tumor). Preoperative pathologic diagnosis by fine-needle aspiration (FNA) was tried in 35 patients (55.6%). Benign SFTP or fibrous tumor with benign nature was found in 15 cases, and malignant SFTP or malignant cell–containing tumors that was not definitely diagnosed as malignant SFTP was found in 13. The remaining seven cases showed fibrosis, blood clots with fibrous tissue debris, or nonspecific inflammation. Evaluation modalities for the follow-up included standard chest radiography, chest computed tomography, and additional abdominal or brain computed tomography.

Resection was performed through a thoracotomy in 37 cases (58.7%), a video-assisted thoracoscopy in 22 cases (34.9%), and a sternotomy in 4 cases (6.4%). Mass excision only at its implantation was performed in 34 cases, but en bloc resection including surrounding structures was performed in the remainder owing to invasion or severe peritumoral inflammatory adhesion (Table 1).

Complete resection was performed in all cases except two. There was no operative morbidity or mortality. The mean follow-up time was 25.9 months. There were 44 (69.8%) pathologically benign SFTP cases, and 19 (30.2%) malignant SFTP cases. The mean follow-up time for benign SFTP was 29.8 months and that for malignant SFTP was 18.7 months (p = 0.01). Local recurrence occurred in three cases. Two of 3 patients were treated by surgical reresection. Distant metastasis after curative resection occurred in eight cases; the metastatic sites were bone (n = 2), brain (n = 2), lung (n = 2), and an intraabdominal lymph node (n = 2). Eight patients died, and they all had malignant SFTP. All of their causes of death were cancer related. All of the benign SFTP patients were alive. Median survival of malignant SFTP patients was 24 months (Fig 1).



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  		Fig 1. Survival after complete resection of solitary fibrous tumor of the pleura. Median survival of malignant (group M) solitary fibrous tumor of the pleura was 24 months. All of the patients with benign (group B) solitary fibrous tumor of the pleura were alive (p < 0.0001). (fu = follow-up.)

 
To identify clinical or radiologic characteristics capable of predicting the pathologic type or clinical outcome, we examined differences in mass size, the proportion of symptomatic patients, and the most favorable impression of the radiologist based on chest computed tomography in malignant and benign cases (Fig 2, Table 2). No statistically significant difference in mass size was observed between the two groups, although the mean size of the malignant group was larger. The proportion of symptomatic patients was higher in the malignant group (p = 0.012). The radiologist's impression of nonpleural tumor (ie, pulmonary mass, mediastinal mass) was more prevalent in the malignant SFTP group, and this was statistically significant.



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  		Fig 2. Chest computed tomographic scan of malignant solitary fibrous tumor of the pleura. This picture shows a very large mass shifting the mediastinum with loculated pleural effusion.

 

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  		Table 2. Correlation Between Clinical Characteristic and Pathologic Type
 
Because decisions on operative approach were mainly determined by preoperative imaging studies in the majority of thoracotomy cases, we studied the correlation between the approach used and the radiologist's impression; however, we found nothing of significance (Table 3).


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  		Table 3. Correlation Between Radiologist's Impression and Type of Operative Approacha
 
When we compared cases that received mass excision only to those that received en bloc resection, the proportion of symptomatic patients was found to be higher in the en bloc resection group (Table 4). The number of cases giving the impression of a pleura including SFTP was greater among mass excision only cases.


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  		Table 4. Correlation Between Extent of Resection and Clinical Variables
 

   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
References
 
Solitary fibrous tumor of the pleura is a rare neoplasm that accounts for 8% of benign pathologic diseases of the chest and 10% of pleural neoplasms [1]. Its incidence is 2.8 per 100,000 registered hospital patients [13]. Approximately 800 cases of SFTP have been reported in the literature between 1931 and 2002 [2]. A slight female preponderance or an even sex distribution, greatest occurrence in the fourth to sixth decades, and no history of asbestos exposure are common points of the present study and in the literature.

Solitary fibrous tumors of the pleura can present with various kinds of symptoms such as intrathoracic symptoms (dyspnea, chest pain, hemoptysis), systemic symptoms (hypoglycemia, hypertrophic osteoarthropathy), or nonspecific symptoms (fever, weight loss, fatigue). The proportion of symptomatic patients has been reported to be 50% to 60%, which is similar to the results of this study. As shown in Tables 3 and 4, the proportion of symptomatic patients is larger in cases of malignant SFTP and en bloc resection. Possible causes may be (1) irritation of adjacent structures by invasion or peritumoral adhesion, or (2) the paracrine action of unknown factors secreted by the SFTP. Moreover, presenting symptoms may help predict the clinical course after a thorough medical history taking and physical examination.

Preoperative pathologic diagnosis (FNA) was performed in 55% of the enrolled patients. In the present study, according to the radiologist's impressions, the FNA group was indistinguishable from the non-FNA group (patients who had undergone resection without preoperative pathologic diagnosis). Furthermore, the radiologist's impressions based on imaging studies were of a benign or malignant pulmonary mass, thymoma, neurogenic tumors, esophageal submucosal tumor, or chronic empyema, all of which ultimately require surgical resection. Because surgical resection involves simultaneous diagnosis and treatment, preoperative FNA should not always be considered. Surgical resection for diagnosis and treatment is acceptable only if the patient is operable, because operative morbidity and mortality are very small. At least repeated FNA is not required even when a definitive preoperative pathologic result is not obtained.

The choice of surgical approach is affected by location of tumors and by spatial relations in the imaging study rather than by the radiologist's impressions (Table 3). In sternotomy cases, impressions on imaging study were thymoma (benign or invasive) or other anterior mediastinal mass. Therefore, the locations of tumors and the surgeon's preference are considered more decisive than the radiologist's impression in terms of the choice of the surgical approach.

The radiologist's impressions are considered related to surgical extent. The impressions of nonpleural tumor were more prevalent among en bloc resection cases. One of the possible causes of this finding may be the atypical images produced by a malignant SFTP showing invasion, severe peritumoral adhesion, necrosis, or hemorrhage. These atypical images differ from images of typical, well-circumscribed pleural tumor. If an SFTP originating from the visceral pleural fold at the interlobar fissure shows such atypical findings, it may more resemble a malignant pulmonary mass than a pleural tumor [14, 15]. Considering the finding that the proportion of patients with the impression of a pleural tumor was higher in the mass excision only group, the radiologist's impression strongly favoring SFTP could be a means of predicting surgical extent preoperatively.

The reported incidence of malignant SFTP varies from 7% to 60%, a variation attributed to slight institutional pathologic criteria differences. Although some authors advocate that the number of mitoses is the most significant criterion among the four criteria of malignant SFTP, useful immunohistochemical prognostic markers of malignant SFTP are still being investigated.

Perrot and colleagues [2] classified SFTP as benign pedunculated, benign sessile, malignant pedunculated, and malignant sessile, ie, a classification on the basis of a combination of gross morphologic features and pathologic type. They reported significant differences in the recurrences and survivals of these morphopathologic subtypes. Recurrences were reported to occur in 63% of those in the malignant sessile group, in 14% in the malignant pedunculated group, in 8% in the benign sessile group, and in 2% of those in the benign pedunculated group. From the patients enrolled in the present study, morphopathologic information could be obtained in 45 cases; 18 were benign sessile cases, 16 benign pedunculated, 10 malignant sessile, and 1 malignant pedunculated. Local recurrence or distant metastasis occurred only in the malignant sessile type. Although the result is not absolutely the same as that of Perrot and associates [2], the pathologically malignant or the sessile forms are considered to be related to a poor prognosis. Prognostic significance of this morphopathologic classification should be prospectively investigated in larger groups.

The treatment of choice for benign SFTP is complete surgical resection. However, as far as malignant SFTP cases are concerned, there is no established systemic therapy, either preoperatively or postoperatively, despite the fact that malignant SFTP has shown distant metastasis [8, 16]. Because a small number of malignant SFTP patients showed survival times of more than 40 months in the present study, preoperative or postoperative systemic therapy should be considered in selected patients who are predicted to achieve a satisfactory result [17]. Thus further study should be performed to identify those factors affecting therapeutic response.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

  1. Klemperer P, Rabin LB. Primary neoplasms of the pleura: a report of five cases Arch Pathol 1931;11:385-412.
  2. Perrot M, Fischer S, Brundler MA, Sekine Y, Keshavjee S. Solitary fibrous tumors of the pleura Ann Thorac Surg 2002;74:285-293.[Abstract/Free Full Text]
  3. Hernandez FJ, Hernandez BB. Localized fibrous tumors of the pleura: a light and electron microscopic study Cancer 1974;34:1667-1674.[Medline]
  4. Al-Azzi M, Thurlow NP, Corrin B. Pleural mesothelioma of connective tissue type, localized fibrous tumor of the pleura, and reactive submesothelial hyperplasia: an immunohistochemical comparison J Pathol 1989;158:41-44.[Medline]
  5. Hanau CA, Miettinen M. Solitary fibrous tumor: histological and immunohistochemical spectrum of benign and malignant variants presenting at different sites Hum Pathol 1995;26:440-449.[Medline]
  6. Suster S, Nascimento AG, Miettinen M, Sickel JZ, Moran CA. Solitary fibrous tumors of the soft tissue: a clinicopathologic and immunohistochemical study of 12 cases Am J Surg Pathol 1995;19:1257-1266.[Medline]
  7. Hasegawa T, Hirose T, Seki K, Yang P, Sano T. Solitary fibrous tumors of the soft tissue: an immunohistochemical and ultrastructural study Am J Clin Pathol 1996;106:325-331.[Medline]
  8. Perrot M, Kurt AM, Robert JH, Borisch B, Spiliopoulos A. Clinical behavior of solitary fibrous tumors of the pleura Ann Thorac Surg 1999;67:1456-1459.[Abstract/Free Full Text]
  9. Rena O, Filosso PL, Papilia E, et al. Solitary fibrous tumor of the pleura: surgical treatment Eur J Cardiothorac Surg 2001;19:185-189.[Abstract/Free Full Text]
  10. Magdeleinat P, Alifano M, Petino A, et al. Solitary fibrous tumors of the pleura: clinical characteristics, surgical treatment and outcome Eur J Cardiothorac Surg 2002;21:1087-1093.[Abstract/Free Full Text]
  11. Nonaka M, Kadokura M, Takaba T. Benign solitary fibrous tumor of the parietal pleura which invaded the intercostal muscle Lung Cancer 2001;31:325-329.[Medline]
  12. Shaker W, Meatchi T, Dusser D, Riquet M. An unusual presentation of solitary fibrous tumor of the pleura: right atrium and inferior vena cava compression Eur J Cardiothorac Surg 2002;22:640-642.[Abstract/Free Full Text]
  13. Okike N, Bernatz E, Woolner B. Localized mesothelioma of the pleura J Thorac Cardiovasc Surg 1978;75:363-372.[Abstract]
  14. Briselli M, Mark EJ, Dickersin GR. Solitary fibrous tumors of the pleura: eight new cases and review of 360 cases in the literature Cancer 1981;47:2678-2689.[Medline]
  15. Aufiero TX, McGary SA, Campbell DB, Phillips PP. Intrapulmonary benign fibrous tumors of the pleura J Thorac Cardiothorac Surg 1995;110:549-551.[Free Full Text]
  16. Carter D, Otis CN. Three types of spindle cell tumors of the pleura: fibroma, sarcoma, and sarcomatoid mesothelioma Am J Surg Pathol 1988;12:747-753.[Medline]
  17. Yokoi T, Tsuzuki T, Yatabe Y, Suzuki M, Kurumaya H, Koshikawa T. Solitary fibrous tumour: significance of p53 and CD34 immunoreactivity in its malignant transformation Histopathology 1998;32:423-432.[Medline]



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