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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="para0002" class="elsevierStylePara elsevierViewall">Primary pleural lymphoma is a rare disease&#44; with few cases reported in literature and corresponding to 2&#46;4&#37; of the primary thoracic wall tumors&#46; Diffuse large B cell lymphoma &#40;DLBCL&#41; is the most common subtype reported&#44; accounting for approximately 30&#37; of non-Hodgking&#39;s lymphomas&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a> Two types of primary pleural lymphomas were originally described in 1987&#44; namely primary effusion lymphoma&#44; principally associated to human immunodeficiency virus &#40;HIV&#41;&#44; and pyothorax-associated lymphoma&#44; more related to human herpes virus-8 &#40;HHV-8&#41; infection and Ebstein-Barr virus &#40;EBV&#41; infection&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0003"><span class="elsevierStyleSup">3</span></a> Pathogenesis has not yet been elucidated but may be related to chronic inflammation of pleura surface that stimulates B-cells lymphocytes uncontrolled proliferation&#44; resulting in pleural lymphoma development&#46; Underlying pleural disease&#44; such as tuberculous pyothorax or pneumothorax&#44; is one of the suggested causes&#46; Other reported cases are associated with some degree of immunosuppression and autoimmune diseases&#44; as vasculitis&#44; or even previous thoracic trauma&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a> Clinical manifestations are unspecific&#44; as the patient may present pleuritic chest pain&#44; dyspnea&#44; cough&#44; fever&#44; anorexia and weigh loss&#46;<a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a> The diagnosis is based on histopathological evidence&#44; using thoracic surgery or thoracoscopy&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">5</span></a> The first choice of treatment is currently in discussion&#44; but chemotherapy is the most frequently used&#46;<a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a> Anthracycline-based chemotherapies combined with anti-CD20 monoclonal antibody rituximab&#44; as cyclophosphamide&#44; pirarubicin&#44; vincristine and prednisolone &#40;CHOP&#41; can significantly improve survival&#44; with complete response rate of 30-45&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0006"><span class="elsevierStyleSup">6</span></a> Cases of spontaneous remission are extremely rare&#44; occurring with complete or partial resolution of the tumor without any treatment&#46; The spontaneous remission mechanisms remain uncharacterized&#46; A possible mechanism is the performance of pleural biopsy that generates a microtrauma and leads to an activation of pro-inflammatory state&#44; alongside with enhanced immune recognition contributing to tumor control&#46;<a class="elsevierStyleCrossRef" href="#bib0007"><span class="elsevierStyleSup">7</span></a></p><p id="para0003" class="elsevierStylePara elsevierViewall">The authors report a clinical case of a 52-year-old female&#44; smoker of 40 pack-years&#44; teacher&#44; with exposure to asbestos in her workplace&#46; The patient had history of osteoporosis and three episodes of pneumonia in adolescence&#46; No history of tuberculosis &#40;TB&#41; or recent contacts with TB&#46; No previous thoracic trauma&#46; No chronic medication&#46; She was referred to pulmonology consultation due to one-month evolution of right posterior pleuritic chest pain which was progressively worsening&#44; with no relief factors&#46; She had no dyspnea&#44; cough&#44; fever or constitutional symptoms&#46; Chest computed tomography &#40;CT&#41; scan revealed macronodular pleural thickening in lower half of right hemithorax with contrast hyperenhancement&#44; associated to moderate right pleural effusion&#59; lymph nodes in azygos-esophageal recess&#44; the largest one with 17 mm diameter&#44; and centrilobular emphysema in upper lobes &#40;<a class="elsevierStyleCrossRef" href="#fig0001">Fig&#46; 1</a>&#41;&#46; Blood count revealed normocytic normochromic anemia &#40;hemoglobin 11&#46;6 g&#47;dL&#44; MGV 87&#46;3 fL&#44; MCH 31&#46;4 pg&#41;&#46; Auto-immune study was normal&#46; There was no evidence of HIV infection&#46; Respiratory functional study was normal&#46; Transthoracic needle aspiration biopsy of pleural nodules was performed&#44; and histopathological exam revealed diffuse pattern of malignant neoplasm extensively occupying the pleural surface&#44; morphologically and immunophenotypically compatible with DLBCL&#46; Microbiology analysis of pleural biopsy was negative&#44; namely Mycobacterium tuberculosis &#40;MTB&#41;&#46; EBV-encoded ribonucleic acid was also tested in the biopsy&#44; and it was negative&#46; Positron emission tomography &#40;PET&#41; scan showed slight uptake of fluor-18-fluorodesoxyglucose &#40;FDG&#41; in pleural nodules and pleural effusion&#44; with no other alterations &#40;<a class="elsevierStyleCrossRef" href="#fig0002">Fig&#46; 2</a>&#41;&#46; Given the diagnostic doubt and the awareness of a rare entity&#44; a surgical biopsy was performed seven weeks later&#46; During the surgery&#44; there was no evidence of pleural effusion and only a 3 mm nodular area was found and biopsied&#44; showing pleural fragments extensively occupied by malignant neoplasia&#46; Immunohistochemical study was positive for CD20&#44; bcl-2&#44; bcl-6 and MUM-1&#44; and negative for CD10&#46; These findings agreed with DLBCL diagnosis&#46; The patient was referred to hematology consultation and the PET scan was repeated three months later&#44; showing total absence of uptake FDG lesions&#46; The patient improved clinically&#44; with no chest pain and normalization of blood count&#46; Complete spontaneous remission of primary pleural lymphoma was admitted&#46; The patient is currently on clinical surveillance without recurrence of the disease after one year of follow-up&#44; confirmed by PET scan&#46;</p><elsevierMultimedia ident="fig0001"></elsevierMultimedia><elsevierMultimedia ident="fig0002"></elsevierMultimedia><p id="para0004" class="elsevierStylePara elsevierViewall">Pleural thickening or nodules associated with pleural effusion raised the hypothesis of an underlying neoplasm&#44; namely pleural metastatic tumors or pleural mesothelioma&#44; especially when exposures to asbestos is found&#44; as in the present clinical case&#46; Primary pleural DLBCL is a rare differential diagnosis of pleural nodules&#44; particularly when there is no previous history of pleural inflammation&#44; such as pneumothorax or pyothorax&#44; immunosuppression&#44; viral infection &#40;HIV&#44; EBV or HHV-8&#41; or thoracic trauma&#46; Clinical examinations such as imaging features and laboratory examination are unspecific&#46; Therefore&#44; an accurate histopathological diagnosis based on thoracic surgery or thoracoscopy is essential&#46;</p><p id="para0005" class="elsevierStylePara elsevierViewall">In conclusion&#44; primary pleural lymphoma is a rare entity principally associated with chronic inflammatory stimulus&#46; Chemotherapy is the most widely used treatment&#44; with complete remissions rates of around 35&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> On the other hand&#44; 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Letter to the Editor
Primary pleural lymphoma – A rare diagnosis
S.S. Guerraa,
Corresponding author
7869@hstviseu.min-saude.pt

Corresponding author.
, S. Coutinhob, T. Abrantesa, P. Rochab, H.M. Silvab
a Serviço de Pneumologia, Centro Hospitalar Tondela-Viseu, Avenida Rei Dom Duarte, 3504-509, Viseu, Portugal
b Serviço de Hematologia, Centro Hospitalar Tondela-Viseu, Avenida Rei Dom Duarte, 3504-509, Viseu, Portugal
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resulting in pleural lymphoma development&#46; Underlying pleural disease&#44; such as tuberculous pyothorax or pneumothorax&#44; is one of the suggested causes&#46; Other reported cases are associated with some degree of immunosuppression and autoimmune diseases&#44; as vasculitis&#44; or even previous thoracic trauma&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a> Clinical manifestations are unspecific&#44; as the patient may present pleuritic chest pain&#44; dyspnea&#44; cough&#44; fever&#44; anorexia and weigh loss&#46;<a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a> The diagnosis is based on histopathological evidence&#44; using thoracic surgery or thoracoscopy&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">5</span></a> The first choice of treatment is currently in discussion&#44; but chemotherapy is the most frequently used&#46;<a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a> Anthracycline-based chemotherapies combined with anti-CD20 monoclonal antibody rituximab&#44; as cyclophosphamide&#44; pirarubicin&#44; vincristine and prednisolone &#40;CHOP&#41; can significantly improve survival&#44; with complete response rate of 30-45&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0006"><span class="elsevierStyleSup">6</span></a> Cases of spontaneous remission are extremely rare&#44; occurring with complete or partial resolution of the tumor without any treatment&#46; The spontaneous remission mechanisms remain uncharacterized&#46; A possible mechanism is the performance of pleural biopsy that generates a microtrauma and leads to an activation of pro-inflammatory state&#44; alongside with enhanced immune recognition contributing to tumor control&#46;<a class="elsevierStyleCrossRef" href="#bib0007"><span class="elsevierStyleSup">7</span></a></p><p id="para0003" class="elsevierStylePara elsevierViewall">The authors report a clinical case of a 52-year-old female&#44; smoker of 40 pack-years&#44; teacher&#44; with exposure to asbestos in her workplace&#46; The patient had history of osteoporosis and three episodes of pneumonia in adolescence&#46; No history of tuberculosis &#40;TB&#41; or recent contacts with TB&#46; No previous thoracic trauma&#46; No chronic medication&#46; She was referred to pulmonology consultation due to one-month evolution of right posterior pleuritic chest pain which was progressively worsening&#44; with no relief factors&#46; She had no dyspnea&#44; cough&#44; fever or constitutional symptoms&#46; Chest computed tomography &#40;CT&#41; scan revealed macronodular pleural thickening in lower half of right hemithorax with contrast hyperenhancement&#44; associated to moderate right pleural effusion&#59; lymph nodes in azygos-esophageal recess&#44; the largest one with 17 mm diameter&#44; and centrilobular emphysema in upper lobes &#40;<a class="elsevierStyleCrossRef" href="#fig0001">Fig&#46; 1</a>&#41;&#46; Blood count revealed normocytic normochromic anemia &#40;hemoglobin 11&#46;6 g&#47;dL&#44; MGV 87&#46;3 fL&#44; MCH 31&#46;4 pg&#41;&#46; Auto-immune study was normal&#46; There was no evidence of HIV infection&#46; Respiratory functional study was normal&#46; Transthoracic needle aspiration biopsy of pleural nodules was performed&#44; and histopathological exam revealed diffuse pattern of malignant neoplasm extensively occupying the pleural surface&#44; morphologically and immunophenotypically compatible with DLBCL&#46; Microbiology analysis of pleural biopsy was negative&#44; namely Mycobacterium tuberculosis &#40;MTB&#41;&#46; EBV-encoded ribonucleic acid was also tested in the biopsy&#44; and it was negative&#46; Positron emission tomography &#40;PET&#41; scan showed slight uptake of fluor-18-fluorodesoxyglucose &#40;FDG&#41; in pleural nodules and pleural effusion&#44; with no other alterations &#40;<a class="elsevierStyleCrossRef" href="#fig0002">Fig&#46; 2</a>&#41;&#46; Given the diagnostic doubt and the awareness of a rare entity&#44; a surgical biopsy was performed seven weeks later&#46; During the surgery&#44; there was no evidence of pleural effusion and only a 3 mm nodular area was found and biopsied&#44; showing pleural fragments extensively occupied by malignant neoplasia&#46; Immunohistochemical study was positive for CD20&#44; bcl-2&#44; bcl-6 and MUM-1&#44; and negative for CD10&#46; These findings agreed with DLBCL diagnosis&#46; The patient was referred to hematology consultation and the PET scan was repeated three months later&#44; showing total absence of uptake FDG lesions&#46; The patient improved clinically&#44; with no chest pain and normalization of blood count&#46; Complete spontaneous remission of primary pleural lymphoma was admitted&#46; The patient is currently on clinical surveillance without recurrence of the disease after one year of follow-up&#44; confirmed by PET scan&#46;</p><elsevierMultimedia ident="fig0001"></elsevierMultimedia><elsevierMultimedia ident="fig0002"></elsevierMultimedia><p id="para0004" class="elsevierStylePara elsevierViewall">Pleural thickening or nodules associated with pleural effusion raised the hypothesis of an underlying neoplasm&#44; namely pleural metastatic tumors or pleural mesothelioma&#44; especially when exposures to asbestos is found&#44; as in the present clinical case&#46; Primary pleural DLBCL is a rare differential diagnosis of pleural nodules&#44; particularly when there is no previous history of pleural inflammation&#44; such as pneumothorax or pyothorax&#44; immunosuppression&#44; viral infection &#40;HIV&#44; EBV or HHV-8&#41; or thoracic trauma&#46; Clinical examinations such as imaging features and laboratory examination are unspecific&#46; Therefore&#44; an accurate histopathological diagnosis based on thoracic surgery or thoracoscopy is essential&#46;</p><p id="para0005" class="elsevierStylePara elsevierViewall">In conclusion&#44; primary pleural lymphoma is a rare entity principally associated with chronic inflammatory stimulus&#46; Chemotherapy is the most widely used treatment&#44; with complete remissions rates of around 35&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> On the other hand&#44; spontaneous remissions are extremely rare&#46;<a class="elsevierStyleCrossRef" href="#bib0007"><span class="elsevierStyleSup">7</span></a> This case highlights the importance of primary pleural lymphoma in differential diagnosis of pleural disease&#44; reinforcing the value of an accurate histological diagnosis&#46;</p><span id="sec0001" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0002">Contributions of the authors</span><p id="para0006" class="elsevierStylePara elsevierViewall">SSG and SC prepared the manuscript&#46; TA and PR were responsible for patient management and revised the manuscript&#46; HMS critically revised the manuscript and approved the final version&#46; All authors read and approved the final manuscript&#46;</p></span></span>"
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