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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="para0002" class="elsevierStylePara elsevierViewall">The simultaneous occurrence of sarcoidosis and lymphoproliferative disease is a rare but well-established entity called sarcoidosis-lymphoma syndrome&#46; Despite being increasingly recognized&#44; it remains a diagnostic challenge due to clinical and imaging similarities&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a> In most cases&#44; sarcoidosis precedes lymphoma&#44; with non-Hodgkin&#39;s lymphoma being the most commonly associated type&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> In this case report we describe a rare presentation of endobronchial Hodgkin lymphoma in a patient with sarcoidosis-lymphoma syndrome&#46;</p><p id="para0003" class="elsevierStylePara elsevierViewall">A thirty-seven-year-old male&#44; non-smoker&#44; was diagnosed with lymph node&#44; bone and cutaneous sarcoidosis in 2018&#44; initially treated with prednisolone 40mg&#47;day&#44; later tapered to a maintenance dose of 10mg&#47;day&#59; and with Stage IV classic Hodgkin&#39;s Lymphoma &#40;HL&#41;&#44; in 2019&#44; treated with ABVD &#40;doxorubicin&#44; bleomycin&#44; vinblastine and dacarbazine&#41; and achieving complete remission in 2020&#46; He was asymptomatic to date&#46;</p><p id="para0004" class="elsevierStylePara elsevierViewall">In February 2021 the patient started to complain of dry cough&#44; fever and night sweats&#46; Physical examination was unremarkable&#46; Laboratory investigations revealed worsening microcytic anemia &#40;hemoglobin of 8&#46;0g&#47;dL&#41; and both high erythrocyte sedimentation rate &#40;72mm&#47;h&#41; and C-reactive protein &#40;11mg&#47;dL&#41;&#46; Chest radiograph was normal&#46;</p><p id="para0005" class="elsevierStylePara elsevierViewall">18-FDG Positron emission tomography was performed and showed multiple supra and infradiaphragmatic foci with active metabolism &#40;elevated maximum standardized uptake values &#40;SUVmax&#41;&#41; suggesting active lymphoproliferative disease&#46;</p><p id="para0006" class="elsevierStylePara elsevierViewall">As there was no biopsiable peripheral lymphadenopathy&#44; the patient underwent endobronchial ultrasound &#40;EBUS&#41;&#46; The procedure revealed multiple pearly nodular lesions throughout the tracheobronchial tree &#40;<a class="elsevierStyleCrossRef" href="#fig0001">Figs&#46; 1</a> and <a class="elsevierStyleCrossRef" href="#fig0002">2</a>&#41;&#46; Bronchial biopsies were performed and mediastinal lymph nodes were punctured at stations 4R and 7 by EBUS-transbronchial needle aspiration&#46;</p><elsevierMultimedia ident="fig0001"></elsevierMultimedia><elsevierMultimedia ident="fig0002"></elsevierMultimedia><p id="para0007" class="elsevierStylePara elsevierViewall">The anatomopathology of the endobronchial lesions showed HL infiltration and the examination of the lymph nodes showed non-caseating epithelioid granulomas compatible with sarcoidosis&#46; HL relapse was assumed&#46;</p><p id="para0008" class="elsevierStylePara elsevierViewall">The patient was started on chemotherapy treatment with protocol DHAP &#40;dexamethasone&#44; cytarabine&#44; cisplatin and prednisolone&#41;&#44; having completed five cycles&#44; with an intermediate response&#46; On reassessment in January 2022&#44; there were signs of disease progression with PET-CT scan showing splenic&#44; multifocal osteomedullary and supra and infra-diaphragmatic lymph nodes involvement&#46; Excisional lymph node biopsy of cervical adenomegaly was performed which&#44; again&#44; confirmed relapse of Hodgkin&#39;s Lymphoma and the patient was started on 3rd-line chemotherapy with BV-GVD &#40;brentuximab vedotin&#160;&#43;&#160;gemcitabine&#44; vinorelbine and doxorubicin&#41; and was proposed for autologous stem cell transplantation&#46;</p><p id="para0009" class="elsevierStylePara elsevierViewall">The sarcoidosis-lymphoma syndrome was first described in 1986 by Brincker&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a></p><p id="para0010" class="elsevierStylePara elsevierViewall">Lymphoproliferative disease develops relatively frequently in patients with chronic active sarcoidosis&#44; probably as a consequence of immunological abnormalities observed in this pathology&#44; such as an increased number of T-helper cells in granulomatous tissues&#44; a decreased number of circulating T-helper cells and hyperactivity of the B-cell system&#46; The prolonged immunosuppression therapy&#44; may also have an important role&#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></p><p id="para0011" class="elsevierStylePara elsevierViewall">The diagnosis of sarcoidosis-lymphoma syndrome is a challenge&#44; since granulomatous inflammation can occur in infections and neoplasms&#44; and is not&#44; in isolation&#44; a diagnosis of sarcoidosis&#46; In our patient&#44; other analytical changes&#44; such as the increase in angiotensin-converting enzyme &#40;ACE&#41; and hypercalcemia&#44; helped the initial diagnosis of sarcoidosis&#46;</p><p id="para0012" class="elsevierStylePara elsevierViewall">The diagnosis of lymphoma was made later by histologic examination of lymph nodes&#44; thus allowing the diagnosis of sarcoidosis-lymphoma syndrome&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> A rare aspect of this case is the association between Hodgkin lymphoma and sarcoidosis&#44; which&#44; despite being less common&#44; is also described in the literature&#46;</p><p id="para0013" class="elsevierStylePara elsevierViewall">Another unusual aspect that is highlighted in this clinical case is the recurrence of lymphoma with a rare endobronchial presentation&#46; Pulmonary involvement of HL may occur in up to 40&#37; of cases&#44; but endobronchial involvement is rare&#44; or may be underdiagnosed&#46;<a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">5</span></a> Its frequency varies according to the authors &#40;described in living patients between 1&#46;9-14&#37; of patients with HL&#41;&#46; The frequency of endobronchial lymphoma is higher post-mortem&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">5</span></a> The secondary bronchi and bronchioles are most often affected&#44; in relation to the main bronchi and trachea&#46; The origin physiopathology is thought to involve transmural spread of hilar adenopathies or blood spread from an extrathoracic focus&#46;<a class="elsevierStyleCrossRef" href="#bib0006"><span class="elsevierStyleSup">6</span></a> Lesions may present as platyform mucosal infiltrates with superficial ulceration or&#44; more rarely&#44; a single polypoid mass&#46;</p><p id="para0014" class="elsevierStylePara elsevierViewall">In this patient&#44; the diagnosis of endobronchial lymphoma was an incidental finding during EBUS procedure aimed to evaluate mediastinal lymphadenopathy&#46; The presence of non-caseating epithelioid granulomas could be inferred as a recurrence of sarcoidosis&#46;</p><p id="para0015" class="elsevierStylePara elsevierViewall">In summary&#44; this case highlights the differential diagnosis between sarcoidosis and lymphoma&#44; the possibility of their overlap and the need for systematic exploration of the tracheobronchial tree in suspected lymphoma&#46; Bronchoscopy evaluation in these patients might allow the histological diagnosis through lesion biopsy and the evaluation of the extent of HL&#44; which will influence the prognosis and treatment of the disease&#46;</p><span id="sec0001" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0002">Authors&#8217; contribution</span><p id="para0016" class="elsevierStylePara elsevierViewall">Study Conception and design&#58; Catarina Barata&#44; Jos&#233; Pedro Bol&#233;o-Tom&#233;&#46;</p><p id="para0017" class="elsevierStylePara elsevierViewall">Data acquisition&#58; Catarina Barata&#44; Margarida Isabel Pereira&#44; Miguel Barbosa&#46;</p><p id="para0018" class="elsevierStylePara elsevierViewall">Data analysis and interpretation&#58; Catarina Barata&#44; Margarida Isabel Pereira&#44; Miguel Barbosa&#44; Filipa Mousinho&#46;</p><p id="para0019" class="elsevierStylePara elsevierViewall">Dafting of the manuscript&#58; Catarina Barata&#44; Margarida Isabel Pereira&#44; Miguel Barbosa</p><p id="para0020" class="elsevierStylePara elsevierViewall">Critical revision of the manuscript for important intelectual content&#58; Filipa Mousinho&#44; Jos&#233; Pedro Bol&#233;o-Tom&#233;&#46;</p><p id="para0021" class="elsevierStylePara elsevierViewall">All authors read and approved the final manuscript&#46;</p></span></span>"
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Letter to the Editor
A curious presentation of an endobronchial Hodgkin's lymphoma in a sarcoidosis-lymphoma syndrome
C. Barataa, M.I. Pereirab, M. Barbosac, F. Mousinhod, J.P. Boléo-Toméb
a Serviço de Pneumologia, Centro Hospitalar de Lisboa Ocidental, EPE, Lisboa, Portugal
b Serviço de Pneumologia, Hospital Professor Doutor Fernando Fonseca, EPE, Amadora, Portugal
c Serviço de Pneumologia, Centro Hospitalar Barreiro Montijo, EPE, Montijo, Portugal
d Serviço de Hematologia, Centro Hospitalar de Lisboa Ocidental, EPE, Lisboa, Portugal
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="para0002" class="elsevierStylePara elsevierViewall">The simultaneous occurrence of sarcoidosis and lymphoproliferative disease is a rare but well-established entity called sarcoidosis-lymphoma syndrome&#46; Despite being increasingly recognized&#44; it remains a diagnostic challenge due to clinical and imaging similarities&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a> In most cases&#44; sarcoidosis precedes lymphoma&#44; with non-Hodgkin&#39;s lymphoma being the most commonly associated type&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> In this case report we describe a rare presentation of endobronchial Hodgkin lymphoma in a patient with sarcoidosis-lymphoma syndrome&#46;</p><p id="para0003" class="elsevierStylePara elsevierViewall">A thirty-seven-year-old male&#44; non-smoker&#44; was diagnosed with lymph node&#44; bone and cutaneous sarcoidosis in 2018&#44; initially treated with prednisolone 40mg&#47;day&#44; later tapered to a maintenance dose of 10mg&#47;day&#59; and with Stage IV classic Hodgkin&#39;s Lymphoma &#40;HL&#41;&#44; in 2019&#44; treated with ABVD &#40;doxorubicin&#44; bleomycin&#44; vinblastine and dacarbazine&#41; and achieving complete remission in 2020&#46; He was asymptomatic to date&#46;</p><p id="para0004" class="elsevierStylePara elsevierViewall">In February 2021 the patient started to complain of dry cough&#44; fever and night sweats&#46; Physical examination was unremarkable&#46; Laboratory investigations revealed worsening microcytic anemia &#40;hemoglobin of 8&#46;0g&#47;dL&#41; and both high erythrocyte sedimentation rate &#40;72mm&#47;h&#41; and C-reactive protein &#40;11mg&#47;dL&#41;&#46; Chest radiograph was normal&#46;</p><p id="para0005" class="elsevierStylePara elsevierViewall">18-FDG Positron emission tomography was performed and showed multiple supra and infradiaphragmatic foci with active metabolism &#40;elevated maximum standardized uptake values &#40;SUVmax&#41;&#41; suggesting active lymphoproliferative disease&#46;</p><p id="para0006" class="elsevierStylePara elsevierViewall">As there was no biopsiable peripheral lymphadenopathy&#44; the patient underwent endobronchial ultrasound &#40;EBUS&#41;&#46; The procedure revealed multiple pearly nodular lesions throughout the tracheobronchial tree &#40;<a class="elsevierStyleCrossRef" href="#fig0001">Figs&#46; 1</a> and <a class="elsevierStyleCrossRef" href="#fig0002">2</a>&#41;&#46; Bronchial biopsies were performed and mediastinal lymph nodes were punctured at stations 4R and 7 by EBUS-transbronchial needle aspiration&#46;</p><elsevierMultimedia ident="fig0001"></elsevierMultimedia><elsevierMultimedia ident="fig0002"></elsevierMultimedia><p id="para0007" class="elsevierStylePara elsevierViewall">The anatomopathology of the endobronchial lesions showed HL infiltration and the examination of the lymph nodes showed non-caseating epithelioid granulomas compatible with sarcoidosis&#46; HL relapse was assumed&#46;</p><p id="para0008" class="elsevierStylePara elsevierViewall">The patient was started on chemotherapy treatment with protocol DHAP &#40;dexamethasone&#44; cytarabine&#44; cisplatin and prednisolone&#41;&#44; having completed five cycles&#44; with an intermediate response&#46; On reassessment in January 2022&#44; there were signs of disease progression with PET-CT scan showing splenic&#44; multifocal osteomedullary and supra and infra-diaphragmatic lymph nodes involvement&#46; Excisional lymph node biopsy of cervical adenomegaly was performed which&#44; again&#44; confirmed relapse of Hodgkin&#39;s Lymphoma and the patient was started on 3rd-line chemotherapy with BV-GVD &#40;brentuximab vedotin&#160;&#43;&#160;gemcitabine&#44; vinorelbine and doxorubicin&#41; and was proposed for autologous stem cell transplantation&#46;</p><p id="para0009" class="elsevierStylePara elsevierViewall">The sarcoidosis-lymphoma syndrome was first described in 1986 by Brincker&#46;<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a></p><p id="para0010" class="elsevierStylePara elsevierViewall">Lymphoproliferative disease develops relatively frequently in patients with chronic active sarcoidosis&#44; probably as a consequence of immunological abnormalities observed in this pathology&#44; such as an increased number of T-helper cells in granulomatous tissues&#44; a decreased number of circulating T-helper cells and hyperactivity of the B-cell system&#46; The prolonged immunosuppression therapy&#44; may also have an important role&#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></p><p id="para0011" class="elsevierStylePara elsevierViewall">The diagnosis of sarcoidosis-lymphoma syndrome is a challenge&#44; since granulomatous inflammation can occur in infections and neoplasms&#44; and is not&#44; in isolation&#44; a diagnosis of sarcoidosis&#46; In our patient&#44; other analytical changes&#44; such as the increase in angiotensin-converting enzyme &#40;ACE&#41; and hypercalcemia&#44; helped the initial diagnosis of sarcoidosis&#46;</p><p id="para0012" class="elsevierStylePara elsevierViewall">The diagnosis of lymphoma was made later by histologic examination of lymph nodes&#44; thus allowing the diagnosis of sarcoidosis-lymphoma syndrome&#46;<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> A rare aspect of this case is the association between Hodgkin lymphoma and sarcoidosis&#44; which&#44; despite being less common&#44; is also described in the literature&#46;</p><p id="para0013" class="elsevierStylePara elsevierViewall">Another unusual aspect that is highlighted in this clinical case is the recurrence of lymphoma with a rare endobronchial presentation&#46; Pulmonary involvement of HL may occur in up to 40&#37; of cases&#44; but endobronchial involvement is rare&#44; or may be underdiagnosed&#46;<a class="elsevierStyleCrossRef" href="#bib0004"><span class="elsevierStyleSup">4</span></a><span class="elsevierStyleSup">&#44;</span><a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">5</span></a> Its frequency varies according to the authors &#40;described in living patients between 1&#46;9-14&#37; of patients with HL&#41;&#46; The frequency of endobronchial lymphoma is higher post-mortem&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">5</span></a> The secondary bronchi and bronchioles are most often affected&#44; in relation to the main bronchi and trachea&#46; The origin physiopathology is thought to involve transmural spread of hilar adenopathies or blood spread from an extrathoracic focus&#46;<a class="elsevierStyleCrossRef" href="#bib0006"><span class="elsevierStyleSup">6</span></a> Lesions may present as platyform mucosal infiltrates with superficial ulceration or&#44; more rarely&#44; a single polypoid mass&#46;</p><p id="para0014" class="elsevierStylePara elsevierViewall">In this patient&#44; the diagnosis of endobronchial lymphoma was an incidental finding during EBUS procedure aimed to evaluate mediastinal lymphadenopathy&#46; The presence of non-caseating epithelioid granulomas could be inferred as a recurrence of sarcoidosis&#46;</p><p id="para0015" class="elsevierStylePara elsevierViewall">In summary&#44; this case highlights the differential diagnosis between sarcoidosis and lymphoma&#44; the possibility of their overlap and the need for systematic exploration of the tracheobronchial tree in suspected lymphoma&#46; Bronchoscopy evaluation in these patients might allow the histological diagnosis through lesion biopsy and the evaluation of the extent of HL&#44; which will influence the prognosis and treatment of the disease&#46;</p><span id="sec0001" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0002">Authors&#8217; contribution</span><p id="para0016" class="elsevierStylePara elsevierViewall">Study Conception and design&#58; Catarina Barata&#44; Jos&#233; Pedro Bol&#233;o-Tom&#233;&#46;</p><p id="para0017" class="elsevierStylePara elsevierViewall">Data acquisition&#58; Catarina Barata&#44; Margarida Isabel Pereira&#44; Miguel Barbosa&#46;</p><p id="para0018" class="elsevierStylePara elsevierViewall">Data analysis and interpretation&#58; Catarina Barata&#44; Margarida Isabel Pereira&#44; Miguel Barbosa&#44; Filipa Mousinho&#46;</p><p id="para0019" class="elsevierStylePara elsevierViewall">Dafting of the manuscript&#58; Catarina Barata&#44; Margarida Isabel Pereira&#44; Miguel Barbosa</p><p id="para0020" class="elsevierStylePara elsevierViewall">Critical revision of the manuscript for important intelectual content&#58; Filipa Mousinho&#44; Jos&#233; Pedro Bol&#233;o-Tom&#233;&#46;</p><p id="para0021" class="elsevierStylePara elsevierViewall">All authors read and approved the final manuscript&#46;</p></span></span>"
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ISSN: 25310437
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