(Chest. 2004;125:1144-1147.)
© 2004
American College of Chest Physicians
Video-Assisted Thoracoscopic Surgery Is a Promising Treatment for Solitary Fibrous Tumor of the Pleura*
Makoto Takahama, MD, PhD;
Keiji Kushibe, MD;
Takeshi Kawaguchi, MD;
Michitaka Kimura, MD and
Shigeki Taniguchi, MD
* From the Department of Thoracic and Cardiovascular Surgery (Drs. Takahama, Kushibe, Kawaguchi, and Taniguchi), Nara Medical University, Kashihara, Nara, Japan; and the Department of Thoracic Surgery (Dr. Kimura), Nara Prefectural Hospital, Nara, Japan.
Correspondence to: Makoto Takahama, MD, PhD, Department of Thoracic and Cardiovascular Surgery, Nara Medical University, 840 Shijo-cho, Kashihara, Nara 634-8522, Japan; e-mail: tmakoto{at}naramed-u.ac.jp
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Abstract
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Study objectives: Solitary fibrous tumors of the pleura (SFTPs) are slow-growing neoplasms. Approximately 800 cases have been reported in the literature to date. The aim of this study was to address our experience with the management of SFTPs and to evaluate the advantage of video-assisted thoracoscopic surgery (VATS) in SFTP treatment.
Design: Retrospective analysis of our experience.
Setting: Department of Thoracic and Cardiovascular Surgery, Nara Medical University.
Patients: From January 1992 to August 2002, 13 patients with a SFTP were referred to us for surgical resection after VATS was adopted in our hospital. The study group consisted of seven men and six women with a mean age of 49.8 years (age range, 37 to 72 years).
Results: Surgical excision was performed with VATS only in nine patients, with VATS plus a small thoracotomy in three patients, and by a posterolateral thoracotomy without VATS in one patient. The mean chest-drain duration was 1.3 days (range, 1 to 3 days), and the mean duration of hospital stay was 8.6 days (range, 3 to 30 days). Eleven tumors, originating from the visceral pleura, were pedunculate, and 2 tumors from the parietal pleura were not pedunculate. Two cases were focally characterized by a mitotic count in excess of four mitoses per 10 high-power fields and by cellular pleomorphism. Follow-up periods ranged from 6 to 120 months, with a mean of 49.7 months and a median of 42 months. All patients have remained well with no recurrence or metastasis.
Conclusions: Complete surgical resection is the treatment of choice for SFTPs. For the pedunculate tumors, therefore, VATS is a powerful and useful approach. Even when it is necessary to perform a small thoracotomy in addition to VATS for the removal of a large tumor, VATS may play an important role in reducing the size of the thoracotomy incision, which results in less invasive surgery. To minimize postoperative morbidity, VATS may be the most promising surgical approach for the resection of SFTPs.
Key Words: pleura • solitary fibrous tumor • video-assisted thoracoscopic surgery
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Introduction
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Primary tumors of the pleura are divided into the following two major categories: diffuse and solitary.1
2
3
Diffuse pleural tumors, which are associated with exposure to asbestos, arise from mesothelial tissue, have poor prognosis, and are more common than solitary pleural tumors.4
5
6
On the other hand, a solitary fibrous tumor of the pleura (SFTP) is a slow-growing neoplasm, with approximately 800 cases having been reported in the literature to date.7
In 1931, Klemper and Rabin8
differentiated a diffuse type of SFTP that arose from the mesothelial layer of a localized form that arose from the mesothelial fibrous connective tissue. SFTPs are usually discovered as asymptomatic lesions on routine chest radiographs in adult patients with no evidence of asbestos exposure. Although they may be relatively large, they are usually treated by simple excision and do not recur if their resection is microscopically complete.9
10
The literature on SFTPs suggests that tumor size, increased mitoses, increased cellularity, increased nuclear pleomorphism, hemorrhage, and necrosis may be associated with clinical behavior.10
11
In addition, some authors describe the presence of a histologically malignant component, with increased cellularity and mitoses, in otherwise benign-appearing SFTPs as a frequent occurrence in tumors that recur or metastasize.9
10
11
12
Recently, the use of video-assisted thoracoscopic surgery (VATS) was introduced for the diagnosis and treatment of intrathoracic lesions. VATS is thought to be minimally invasive and, thus, is perceived to result in less postoperative pain and quicker recovery.13
14
The aim of this study was to summarize our experience in the management of SFTPs and to evaluate the advantage of VATS for the treatment of SFTPs.
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Materials and Methods
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From January 1992 to December 2002, 13 patients with SFTPs were referred for surgical resection at Nara Medical University Hospital after VATS was adopted in our hospital. Our series included seven men and six women with a mean age of 49.8 years (age range, 37 to 72 years) [Table 1
]. The preoperative follow-up period was 2.8 years (range, 1 to 6 years) in the outpatient clinic of other hospitals. The tumor was right-sided in eight patients (61.5%) and left-sided in five patients (38.5%). None of the patients was symptomatic or presented with a history of hypoglycemia, but five of them were associated with diabetes mellitus. No history of exposure to asbestos was recorded in any patient. Ten of the patients were current smokers or ex-smokers. All patients underwent chest roentgenography and CT scanning of the chest. Three patients (23.1%) underwent CT-guided, fine-needle aspiration biopsy.
The diagnosis was confirmed by corroborative morphologic and immunohistochemical findings that are available for review. The pathologic diagnostic criteria of SFTP used were circumscribed tumors characterized by a haphazard growth pattern (so-called patternless pattern or storiform pattern) of short spindle cells with scant cytoplasm and bland cytologic appearance, separated by strands of rope-like collagen. Typically, these tumors exhibit an alteration of hypercellular (ie, collagen-rich) areas.5
6
9
Small to large numbers of blood vessels were observed. The malignant variant was macroscopically similar to the benign tumor, but contained high mitotic activity, which was defined as at least four mitotic figures per 10 high-power fields (HPFs), necrosis, and mild-to-marked pleomorphism, based on nuclear size, irregularity, and the presence of nuclear atypia.5
7
12
15
Clinical information was obtained by means of hospital records and telephone calls to patients.
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Results
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Resection was completed in all our patients. Surgical excision was performed by VATS in nine patients (69.2%), by VATS plus a small thoracotomy (< 5 cm) in three patients (23.1%), and by posterolateral thoracotomy accompanied by VATS in one patient (7.7%) [Table 2
]. Mean chest drain duration was 1.3 days (range, 1 3 days), and the mean hospital stay was 8.6 days (range, 3 to 30 days). The perioperative mortality rate was 0%. At surgery, 11 tumors (84.6%) arising from the visceral pleura were pedunculate, and 2 tumors (15.4%) from the parietal pleura presented with a broad base of attachment (Table 2)
. All tumors were resected with free surgical margins. In two patients, a pleural effusion was observed in the ipsilat-eral thoracic cavity. Grossly, the tumors appeared well-circumscribed, polylobulated, with a smooth sur-face and firm consistency. They measured from 25 x 15 x 15 mm to 120 x 80 x 40 mm. On sectioning, the tumors showed a whorled, fleshy appearance, sometimes alternating with some myxoid areas. Focal necrosis and hemorrhagic zones were found in two cases (15.4%), both relatively large in diameter. Microscopically, all the tumors consisted of spindle-shaped cells. The tumor cells and intercellular collagen fibers proliferated without obvious direction or structure, which corresponded to the so-called "patternless pattern." Two cases (15.4%) were focally characterized by a mitotic count in excess of four mitoses per 10 HPFs and by cellular pleomorphism (Tables 2
and 3
). Immunohistochemical analysis revealed positivity for CD34 and vimentin in all tumors, and no expression of cytokeratin, desmin, S-100, Ki-67, or actin in any of the tumors.
All patients were included in a follow-up program that included clinical examination and chest roentgenogram after 1, 3, and 6 months postoperatively, and annually thereafter. Follow-up took place until December 2002, and ranged from 6 to 120 months in length, with a mean of 49.7 months. All of the patients have remained well, with no recurrence or metastasis of the tumors.
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Discussion
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SFTPs are rare, and their incidence is approximately 2.8 cases of 100,000 registered hospital patients.5
The clinical behavior of SFTPs is not well-known.4
7
16
Although most of them are considered to be histologically benign, local recurrences and enlargement without signs of invasion or metastasis have been reported.1
7
In our series, no recurrence or metastasis of SFTP developed during postoperative mean and median follow-up period of 49.7 and 42 months, respectively. However, two cases showed a mitotic count in excess of four mitoses per 10 HPFs or cellular pleomorphism, both of which are considered to indicate potentially malignant SFTPs.1
12
17
Morphologic indicators also seem to be important predictors of outcome. de Perrot et al7
classified SFTPs into four categories by the morphologic and histologic indicators. In our series, 10 tumors were classified as stage 0, 2 were classified as stage 1, and 1 was classified as stage 2. Even though the risk of recurrence or metastasis is low, some patients have presented with recurrent or metastatic tumors, and therefore careful postoperative observation is required during follow-up of these patients.
In our series, none of the patients presented with a history of hypoglycemia, but five of them were associated with diabetes mellitus. SFTPs can secrete insulin-like growth factor II, which causes refractory hypoglycemia in some patients.18
A high serum level of insulin-like growth factor II is associated with a low level of serum insulin. This finding may relate to the high incidence of the patients with diabetes mellitus.
Immunohistochemical analysis has been an extremely useful tool to differentiate SFTPs from mesotheliomas and other sarcomas.6
17
Indeed, SFTPs, by definition, are vimentin-positive and keratin-negative. In addition, most benign and malignant SFTPs are positive for CD34, whereas CD34 is not present in other pulmonary tumors. The antiapoptotic proto-oncogene, bcl-2, is strongly expressed in SFTPs and is used to differentiate SFTPs from hemangiopericytomas, which are CD34-positive. Occasionally, malignant SFTPs may be CD34-negative.17
19
Although this CD34 negativity in malignant SFTPs may be caused by a dedifferentiation of the tumor and reflects poor prognosis,12
it most likely represents a group of SFTPs that have always been CD34-negative. The expression of bcl-2 can be a useful marker in these CD34-negative tumors to confirm the definitive diagnosis of SFTP.7
12
Complete surgical resection is the treatment of choice for SFTP.4
20
Pedunculate tumors can be safely treated by wedge resection. For these pedunculate tumors, VATS provides a powerful and useful approach. In addition, some authors have reported the assistance of thoracoscopy to obtain a more precise view of the resection margins, even in parietal tumors.1
5
20
Whereas a small thoracotomy can be added to the VATS for the removal of large tumors, VATS plays an important role in reducing the size of the thoracotomy incision, which results in less invasive surgery. Initially, we selected a thoracoscopic approach for the resection of SFTPs in all cases. Even with large tumors, if a VATS resection is technically feasible, it can be carried out. If necessary, we add a small thoracotomy incision to remove the tumor safely and completely, with free surgical margins. Nomori et al20
reported that contact metastasis and local recurrence had occurred at the port site, which was utilized during the VATS procedure. To avoid contact metastasis and local recurrence at the port site, we extract the surgical specimen by means of a retrieval bag. The theoretical advantages of VATS in minimizing postoperative morbidity are appealing. In conclusion, we believe that VATS is the procedure of first choice for the resection of SFTPs.
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Acknowledgements
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We express our gratitude to David K. C. Cooper for his correction of English and careful reading of this manuscript, and to Nami Makimura and Rico Tanaka for editorial assistance.
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Footnotes
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Abbreviations: HPF = high-power field; SFTP = solitary fibrous tumor of the pleura; VATS = video-assisted thoracoscopic surgery
Received for publication July 16, 2003.
Accepted for publication August 29, 2003.
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References
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|---|
- England, DM, Hochholzer, L, McCarthy, MJ (1989) Localized benign and malignant fibrous tumors of the pleura: a clinicopathologic review of 223 cases. Am J Surg Pathol 13,640-658[ISI][Medline]
- 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[CrossRef][ISI][Medline]
- Aufiero, TX, McGary, SA, Campbell, DB, et al Intrapulmonary benign fibrous tumor of the pleura. J Thorac Cardiovasc Surg 1995;110,549-551[Free Full Text]
- Cardillo, G, Facciolo, F, Cavazzana, AO, et al Localized (solitary) fibrous tumors of the pleura: an analysis of 55 patients. Ann Thorac Surg 2000;70,1808-1812[Abstract/Free Full Text]
- Okike, N, Bernatz, PE, Woolner, LB Localized mesothelioma of the pleura: benign and malignant variants. J Thorac Cardiovasc Surg 1978;75,363-372[Abstract]
- van de Rijn, M, Lombard, CM, Rouse, RV Expression of CD34 by solitary fibrous tumors of the pleura, mediastinum, and lung. Am J Surg Pathol 1994;18,814-820[ISI][Medline]
- de Perrot, M, Fischer, S, Brundler, MA, et al Solitary fibrous tumors of the pleura. Ann Thorac Surg 2002;74,285-293[Abstract/Free Full Text]
- Klemper, P, Rabin, CB Primary neoplasm of the pleura: a report of a case. Arch Pathol 1931;11,385-412[ISI]
- Scharifker, D, Kaneko, M Localized fibrous "mesothelioma" of pleura (submesothelial fibroma): a clinicopathologic study of 18 cases. Cancer 1979;43,627-635[CrossRef][ISI][Medline]
- Dalton, WT, Zolliker, AS, McCaughey, WT, et al Localized primary tumors of the pleura: an analysis of 40 cases. Cancer 1979;44,1465-1475[CrossRef][ISI][Medline]
- Witkin, GB, Rosai, J Solitary fibrous tumor of the mediastinum: a report of 14 cases. Am J Surg Pathol 1989;13,547-557[ISI][Medline]
- Gold, JS, Antonescu, CR, Hajdu, C, et al Clinicopathologic correlates of solitary fibrous tumors. Cancer 2002;94,1057-1068[CrossRef][ISI][Medline]
- Li, WW, Lee, TW, Lam, SS, et al Quality of life following lung cancer resection: video-assisted thoracic surgery vs thoracotomy. Chest 2002;122,584-589[Abstract/Free Full Text]
- Roviaro, GC, Varoli, F, Vergani, C, et al State of the art in thoracoscopic surgery: a personal experience of 2000 videothoracoscopic procedures and an overview of the literature. Surg Endosc 2002;16,881-892[CrossRef][ISI][Medline]
- Sanguinetti, CM, Marchesani, F, Ranaldi, R, et al Localized fibrous pleural tumour of the interlobular pleura. Eur Respir J 1996;9,1094-1096[Abstract]
- Robinson, LA, Reilly, RB Localized pleural mesothelioma: the clinical spectrum. Chest 1994;106,1611-1615[Abstract/Free Full Text]
- Krismann, M, Adams, H, Jaworska, M, et al Patterns of chromosomal imbalances in benign solitary fibrous tumours of the pleura. Virchows Arch 2000;437,248-255[CrossRef][ISI][Medline]
- Chamberlain, MH, Taggart, DP Solitary fibrous tumor associated with hypoglycemia: an example of the Doege-Potter syndrome. J Thorac Cardiovasc Surg 2000;119,185-187[Free Full Text]
- Natkunam, Y, Rouse, RV, Zhu, S, et al Immunoblot analysis of CD34 expression in histologically diverse neoplasms. Am J Pathol 2000;156,21-27[Abstract/Free Full Text]
- Nomori, H, Horio, H, Fuyuno, G, et al Contacting metastasis of a fibrous tumor of the pleura. Eur J Cardiothorac Surg 1997;12,928-930[Abstract]
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Abstract
Introduction
