Eur J Cardiothorac Surg 2000;18:491-494
© 2000 Elsevier Science NL
A case of multiple synchronous localized fibrous tumor of the pleura
rfan Ta
tepea,
Ayin Alpera,
Hatice Esra Özayd
nb,
Leyla Memic,
Güven Çetina
a Department of Thoracic Surgery, Atatürk Center For Chest Disease's and Thoracic Surgery, Yüksel Caddesi, 34/15, Kzlay, Ankara, Turkey
b Department of Pathology, Atatürk Center For Chest Disease and Thoracic Surgery, Yüksel Caddesi, 34/15, Kzlay, Ankara, Turkey
c Department of Pathology, Gazi University, Ankara, Turkey
Received 2 February 2000;
received in revised form 5 May 2000;
accepted 23 May 2000.
Corresponding author. Tel.: +90-312-435-1493
e-mail: sumena{at}yahoo.com
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Abstract
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We report a patient with two synchronous distinct masses in the same hemithorax both of which got the diagnosis of benign localized fibrous tumor of the pleura. The plain chest X-ray was rather obscured due to a large left-sided pleural effusion, but her subsequent computerized chest tomography revealed a heterogenous hypodense soft tissue mass, which was pleural in origin, sitting on the diaphragm bathed in fluid. At thoracotomy, we detected two distinct masses in the left hemithorax, both arising from the visceral pleura via their vascular pedicles.
Key Words: Multiple synchronous fibrous tumor • Pleura
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1. Introduction
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Primary pleural tumors have historically been divided into diffuse and localized types [1–5]. The diffuse form, malignant mesothelioma, representing 75–90% of all cases and associated with asbestos exposure, is well known for its rapidly fatal course [6]. Localized fibrous tumor of the pleura (LFTP), is much less common and less well-defined. It is believed to originate from a primitive submesothelial mesencyhmal cells, rather than the mesothelial lining itself [1,2,4,5]. It exists in benign and malignant forms in a ratio of 7:1 [2]. Localized fibrous tumor of the pleura is a unique mass in most cases. Multiple tumors are extremely rare [1,3–5]. In this paper, we report a patient with two synchronous distinct masses in the same hemithorax both of which got the diagnosis of benign LFTP.
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2. Case report
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A 68-year-old woman was referred to our hospital for progressive dyspnea and excessive non-productive cough of a 3 months’ duration, in addition to her night sweats, fatigue and weight loss. Her past medical history was not remarkable except an operation for urinary bladder rupture after a traffic accident 4 years ago. She had prior exposure to asbestos. On physical examination she had dull percussion and absent breath sounds below the level of the left scapula. The plain chest X-ray was rather obscured due to a large left-sided pleural effusion, but her subsequent computerized chest tomography revealed a heterogenous hypodense soft tissue mass, which may be pleural in origin, bathed in fluid (Fig. 1)
. No additional mass could be identified but there were some indirect clues suggesting a mass compression on the paranchyme of the lingular segment. Routine blood analyses were within the reference limits. Bronchoscopy showed extrinsic compression of the lingular segment bronchus, but no endobronchial lesion. Bronchoalveolar lavage cytology revealed no atypical cells and was negative for mycobacteria. A left-sided thoracentesis was performed and 800 ml of pleural fluid was evacuated. The fluid was an exudate with a relatively high glucose level (176 mg/dl) and a high pleural fluid to serum, protein ratio (0.75). Fig. 3 shows the chest X-ray after evacuation of the pleural effusion. It was still difficult to identify the origin of the mass. Consecutive cytological studies of the pleural fluid were negative for any malignity and smears were negative for mycobacteria. A transparietal biopsy was reported to be a mesenchymal tumor of dubious nature by the pathologist.
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Fig. 1. Computed tomographic scan showing a soft tissue mass, may be pleural in origin, bathed in fluid. The arrow indicates the mass.
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At thoracotomy, we detected two distinct masses in the left hemithorax, both arising from the visceral pleura via their vascular pedicles. The larger of the two masses: 15x10x10 cm in size, originated from the diaphragmatic surface of the visceral pleura of the left lower lobe and the smaller; 12x8x4 cm in size, originated from the visceral pleura of the anterior segment of the left upper lobe. They were completely excised with satisfactory margins. Both were well-circumscribed, discoid and lobulated. They were encapsulated with a smooth, bright membrane. The cut surface of the tumors appeared nodular, gray-white in color and commonly firm to rubbery, but had mucoid consistency in rare areas. Histologically the lesions were composed of uniform, collagen-forming spindle cells in haphazardly arranged fascicles (Fig. 2)
. The degree of cellularity varied by location. A low mitotic rate less than one mitosis per ten high power fields was remarkable (Fig. 3
). Immunohistochemical studies for each tumor were strongly positive for vimentin and CD34, but negative for keratin. The pathologic diagnosis of both masses were benign LFTP. Since the parietal pleura was clear and no plaques were detected over its surface, we did not take a sample of parietal pleura. The patient went through an uneventful recovery with regression of her preoperative symptoms.
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Fig. 2. Proliferation of collagen-forming spindle cells arranged in haphazard fascicles (hemotoxylin and eosin, original magnification x40).
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Fig. 3. The chest X-ray after evacuation of the pleural effusion. It was still difficult to identify the origin of the mass.
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3. Discussion
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It has gained common acceptance that a history of asbestos exposure is lacking in patients with LFTP, while it is marked (over 60%) in diffuse malignant mesothelioma [1–5]. One exception is our patient who had a history of asbestos exposure perhaps just as a coincidence but worth remarking.
While an exudative effusion is present in 80% of the patients with diffuse malignant mesothelioma, none of the effusions associating a LFTP has been reported to be an exudate until now [3,6,7]. We have demonstrated an exudative effusion of moderate size in our patient. Her exudate together with her history of asbestos exposure was highly suggestive of a malignant mesothelioma at first sight but her intraoperative findings and pathological examination met all of the criteria for a benign LFTP.
Although LFTP are defined as ‘almost always solitary’ in the literature [4,5], very few occasions of multiple tumors have been reported [1,3,8,9]. Our case has been enrolled in the list which is less than ten in number. A multifocal synchronous tumorogenesis could be due to the exposure to hypothetical predisposing risk factors creating a favorable environment for a multifocal growth as Brunelli et al. [9] suggested. Propagation by contiguity is rather unlikely when repetitive cytological studies (preoperative and intraoperative) of the moderate pleural effusion in our patient revealed no atypical cells, and blood borne metastasis seems almost irrational when benignity was so marked in both of our tumors. Since preoperative diagnosis is difficult to establish, an inevitable thoracotomy is both diagnostic and curative in LFTP provided that the tumor is completely excised [1–3]. Although histologically benign, LFTP may reoccur after surgery or occasionally transform into malignant variants after several years. Five in 78 cases which were diagnosed to be benign histologically at operation had recurrence later in Japan [10]. Therefore complete surgical resection and long term follow-up is recommended for all cases [1–3]. After 12 months’ follow-up, our patient has no signs of recurrence.
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References
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- England D.M., Hochholzer L., McCarthy M.J. Localized benign and malignant fibrous tumors of the pleura. A clinicopathologic review of 223 cases. Am J Surg Pathol 1989;13(8):640-658.[Medline]
- 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]
- Suter M., Gebhard S., Boumghar M., Peloponisios N., Genton C.Y. Localized fibrous tumors of the pleura: 15 new cases and review of the literature. Eur J Cardio-thorac Surg 1998;14(5):453-459.
- Shields T.W. General thoracic surgery, 4th ed 1994:722-731.
- Light R.W. Pleural disease, 3rd ed Baltimore, MD: Williams & Wilkins, 1995:8-17.
- Robinson L.A., Reilly R.B. Localized pleural mesothelioma. The clinical spectrum. Chest 1994;106:1611-1615.[Abstract/Free Full Text]
- Ulrik C.S., Viskum K. Fibrous pleural tumor producing 171 litres of transudate. Eur Respir J 1998;12:1230-1232.[Abstract]
- Urschel J.D., Brooks J.S., Werness B.A., Antkowiak J.G., Takita H. Metachronous benign solitary fibrous tumors of the pleura (localized ‘mesotheliomas’): a case report. Can J Surg 1998;41(6):467-469.[Medline]
- Brunelli A., Sabbaniti A., Catalini G., Fianchini A. Intrapulmonary benign fibrous tumor of the pleura. J Thorac Cardiovasc Surg 1996;111(6):1292-1293.[Free Full Text]
- Hatta T., Ohyabu H., Kurisu S., Tachibana S., Kita Y., Oki K., Okada M., Uematsu M., Matsuda S., Eno K. A case of localized pleural mesothelioma. Kyobu Geka 1991;44(4):344-346.[Medline]
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Abstract
1. Introduction
