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A chest X-ray showed a large retrosternal opacity &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41; and the chest computed tomography &#40;CT&#41; confirmed the presence of a retrosternal lobulated mass&#44; 10&#46;2<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;7<span class="elsevierStyleHsp" style=""></span>cm&#44; in the right hemithorax &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C and D&#41;&#46; A percutaneous CT-guided biopsy was carried out and the histology showed evidence of mesenchymal neoplasm without necrosis&#44; significant pleomorphism or mitotic figures&#46; The tumor cell population stained diffusely for CD 34&#44; vimentin&#44; <span class="elsevierStyleItalic">bcl-2</span> and CD 99 and negative for S-100&#46; Ki-67 was &#60;5&#37;&#46; Although a benign SFTP was diagnosed based on the histology&#44; a right thoracotomy was performed for diagnosis and treatment&#46; A pedunculated tumoural mass was complete resected with segmental resection of the right upper lobe&#46; Immunohistochemistry showed positivity for CD-34 and <span class="elsevierStyleItalic">bcl-2</span> confirming SFTP&#46; The pathological examination revealed multiple mitotic figures &#40;12 mitoses per 10 high-power fields&#41;&#44; mild pleomorphism&#44; hypercellularity&#44; focal hemorrhage and several areas of necrosis and the resection margins were tumor-free&#46; According to these features the tumor was classified as malignant&#46; The post-operative period was uneventful and the patient was discharged home&#46; The patient is now in the third month after surgery without any sign of local recurrence or metastization&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">SFTP is a rare tumor with generally an indolent course and good prognosis with a 10-year survival rate up to 98&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> It occurs mainly in individuals in their sixth or seventh decades of life without gender bias&#46; There is no association with tobacco&#44; asbestos or other exposures&#46; More than 50&#37; of the patients are asymptomatic<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> and the tumor is frequently an incidental finding on a routine chest X-ray&#46; When symptomatic&#44; the patients usually present non-specific respiratory symptoms such as dyspnea&#44; cough and chest pain&#44; and more rarely systemic symptoms&#46; Occasionally&#44; SFTP can manifest as paraneoplastic syndromes&#44; such as hypertrophic pulmonary osteoarthropathy &#40;Pierre Marie-Bamberger syndrome&#41; and refractory hypoglycemia &#40;Doege-Potter syndrome&#41;&#46; The frequency of these syndromes increases with tumor size&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Malignant SFTP is uncommon and its incidence varies from 7&#37; to 60&#37;&#46; It rarely arises from a pre-existing SFTP undergoing malignant transformation&#46; The role of percutaneous CT-guided biopsy is not yet established because its diagnostic accuracy is low<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> and it can underestimate the degree of malignancy given the size and histological heterogeneity of these tumors&#46; Therefore&#44; a pre-surgical diagnosis should not affect the decision to perform surgery for benign and malignant SFTP&#46; According to the world Health Organization Classification for soft tissue tumors&#44; the malignant SFTP is diagnosed if one or more of the following histologic features are present&#58; hypercellularity&#44; pleomorphism&#44; tumor necrosis&#44; more than 4 mitoses per ten high-power fields and infiltrative margins&#46; Immunohistochemistry may be useful in differentiating the SFTP from mesotheliomas and intrapleural sarcomas&#46; Generally SFTP is vimentin&#44; CD34&#44; CD99 and <span class="elsevierStyleItalic">bcl</span>-2 positive and cytokeratin negative&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Complete resection of tumor is the treatment of choice&#44; the only demonstrated effective treatment and the most important prognostic factor&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> The choice of surgical approach &#40;video-assisted thoracoscopic surgery &#40;VATS&#41; and standard thoracotomy&#41; is essentially based on tumor size and the difficulty of removal&#46; The role of adjuvant radio and chemotherapy in malignant SFTP remains unclear since there is no systematic assessment due to the rarity of the tumor&#46; Despite this&#44; there are some reports showing good response to adjuvant radiotherapy in tumors with incomplete resection&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> Recurrence rates for completely resected malignant SFTP range from 14&#37; to 63&#37;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> in pedunculated and sessile tumors&#44; respectively&#44; and occur mainly in the first 24 months after surgery&#46; Despite a complete resection&#44; malignant SFTP can have a poor prognosis with a 5-year rate survival of 45&#44;5&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> Recurrent tumors should be resected and combined chemotherapy with temozolomide and bevacizumab can be considered in locally advanced&#44; recurrent or unresectable malignant SFTP&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">We report a case of a malignant SFTP&#44; a rare pleural tumor that although completely resected and potentially with good prognosis&#44; should have long-term follow-up due to the high risk of local and metastatic recurrence&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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Letter to the Editor
Solitary fibrous tumors of the pleura: not always a benign entity
L.M. Nascimento
Corresponding author
lu_maria_nascimento@hotmail.com

Corresponding author.
, T. Gomes, A. Fernandes, A. Afonso
Department of Pulmonology, Centro Hospitalar de Trás-os-Montes e Alto, Vila Real, Portugal
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A chest X-ray showed a large retrosternal opacity &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41; and the chest computed tomography &#40;CT&#41; confirmed the presence of a retrosternal lobulated mass&#44; 10&#46;2<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;7<span class="elsevierStyleHsp" style=""></span>cm&#44; in the right hemithorax &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C and D&#41;&#46; A percutaneous CT-guided biopsy was carried out and the histology showed evidence of mesenchymal neoplasm without necrosis&#44; significant pleomorphism or mitotic figures&#46; The tumor cell population stained diffusely for CD 34&#44; vimentin&#44; <span class="elsevierStyleItalic">bcl-2</span> and CD 99 and negative for S-100&#46; Ki-67 was &#60;5&#37;&#46; Although a benign SFTP was diagnosed based on the histology&#44; a right thoracotomy was performed for diagnosis and treatment&#46; A pedunculated tumoural mass was complete resected with segmental resection of the right upper lobe&#46; Immunohistochemistry showed positivity for CD-34 and <span class="elsevierStyleItalic">bcl-2</span> confirming SFTP&#46; The pathological examination revealed multiple mitotic figures &#40;12 mitoses per 10 high-power fields&#41;&#44; mild pleomorphism&#44; hypercellularity&#44; focal hemorrhage and several areas of necrosis and the resection margins were tumor-free&#46; According to these features the tumor was classified as malignant&#46; The post-operative period was uneventful and the patient was discharged home&#46; The patient is now in the third month after surgery without any sign of local recurrence or metastization&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">SFTP is a rare tumor with generally an indolent course and good prognosis with a 10-year survival rate up to 98&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> It occurs mainly in individuals in their sixth or seventh decades of life without gender bias&#46; There is no association with tobacco&#44; asbestos or other exposures&#46; More than 50&#37; of the patients are asymptomatic<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> and the tumor is frequently an incidental finding on a routine chest X-ray&#46; When symptomatic&#44; the patients usually present non-specific respiratory symptoms such as dyspnea&#44; cough and chest pain&#44; and more rarely systemic symptoms&#46; Occasionally&#44; SFTP can manifest as paraneoplastic syndromes&#44; such as hypertrophic pulmonary osteoarthropathy &#40;Pierre Marie-Bamberger syndrome&#41; and refractory hypoglycemia &#40;Doege-Potter syndrome&#41;&#46; The frequency of these syndromes increases with tumor size&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Malignant SFTP is uncommon and its incidence varies from 7&#37; to 60&#37;&#46; It rarely arises from a pre-existing SFTP undergoing malignant transformation&#46; The role of percutaneous CT-guided biopsy is not yet established because its diagnostic accuracy is low<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> and it can underestimate the degree of malignancy given the size and histological heterogeneity of these tumors&#46; Therefore&#44; a pre-surgical diagnosis should not affect the decision to perform surgery for benign and malignant SFTP&#46; According to the world Health Organization Classification for soft tissue tumors&#44; the malignant SFTP is diagnosed if one or more of the following histologic features are present&#58; hypercellularity&#44; pleomorphism&#44; tumor necrosis&#44; more than 4 mitoses per ten high-power fields and infiltrative margins&#46; Immunohistochemistry may be useful in differentiating the SFTP from mesotheliomas and intrapleural sarcomas&#46; Generally SFTP is vimentin&#44; CD34&#44; CD99 and <span class="elsevierStyleItalic">bcl</span>-2 positive and cytokeratin negative&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Complete resection of tumor is the treatment of choice&#44; the only demonstrated effective treatment and the most important prognostic factor&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> The choice of surgical approach &#40;video-assisted thoracoscopic surgery &#40;VATS&#41; and standard thoracotomy&#41; is essentially based on tumor size and the difficulty of removal&#46; The role of adjuvant radio and chemotherapy in malignant SFTP remains unclear since there is no systematic assessment due to the rarity of the tumor&#46; Despite this&#44; there are some reports showing good response to adjuvant radiotherapy in tumors with incomplete resection&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> Recurrence rates for completely resected malignant SFTP range from 14&#37; to 63&#37;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> in pedunculated and sessile tumors&#44; respectively&#44; and occur mainly in the first 24 months after surgery&#46; Despite a complete resection&#44; malignant SFTP can have a poor prognosis with a 5-year rate survival of 45&#44;5&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> Recurrent tumors should be resected and combined chemotherapy with temozolomide and bevacizumab can be considered in locally advanced&#44; recurrent or unresectable malignant SFTP&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">We report a case of a malignant SFTP&#44; a rare pleural tumor that although completely resected and potentially with good prognosis&#44; should have long-term follow-up due to the high risk of local and metastatic recurrence&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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ISSN: 21735115
Original language: English
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