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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Bronchogenic cysts are uncommon congenital lesions usually located in the mediastinum or lung and resulting from abnormal budding of foregut in the embryonic stage&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">1&#44;2</span></a> Malignant transformation is very rare and only few well-documented cases have been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">3&#8226;8</span></a> In these cases&#44; histological findings were bronchioalveolar carcinoma&#44; adenocarcinoma&#44; squamous cell carcinoma and large cell carcinoma&#46; To the best of our knowledge&#44; this is the first case of enteric adenocarcinoma arising from an intrapulmonary bronchogenic cyst&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 76-year-old woman with a medical history of hypertension&#44; hypercholesterolemia&#44; hypoacusis&#44; glaucoma&#44; rheumatic polymyalgia and bronchial asthma was referred to our centre for surgical resection of a bronchogenic cyst diagnosed 12 years previously in another hospital&#46; The patient&#44; a farmer&#44; did not have any allergies or toxic habits&#46; She was completely asymptomatic&#44; and physical examination only revealed an absence of breath sounds in the posterior right lower hemithorax&#46; All laboratory tests were normal&#46; The computed tomography &#40;CT&#41; showed a 10&#46;6<span class="elsevierStyleHsp" style=""></span>cm mass in the right lower lobe with calcified walls&#44; which was compatible with bronchogenic cyst &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The bronchoscopy showed an irregular white endobronchial mass at the entrance of the right lower bronchus&#44; which also affected and partially occluded the middle lobe bronchus&#46; The histology of the endobronchial lesion was mucinous adenocarcinoma of the lung&#46; Based on these findings&#44; a PET-CT was performed&#44; which showed an ametabolic mass with a mild increased uptake in the peripheral area and in mediastinal lymph nodes &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The patient underwent functional pulmonary testing and was considered eligible for surgery&#46; She underwent right lower lobectomy with lymph node dissection through muscle-sparing thoracotomy&#46; The postoperative period was uneventful and the patient was discharged 6 days after surgery&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Definitive histology of the specimen was infiltrating colloid adenocarcinoma with immunophenotype of enteric adenocarcinoma arising from a benign cystic mucoid lesion compatible with bronchogenic cyst&#46; Immunohistochemical staining was positive for cytokinin 20 and CDX 2&#44; and negative for cytokinin 20 and TTF1&#46; Mediastinal lymph nodes were not affected&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The final diagnosis was enteric adenocarcinoma arising from a bronchogenic cyst&#46; No additional lesions were found on colonoscopy and further abdominal exams&#46; Because no signs of disease dissemination were found&#44; no additional therapy was applied&#46; The patient is alive without recurrence of disease 6 months after the surgery&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Bronchogenic cysts are congenital lesions thought to originate from the primitive ventral foregut&#44;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">2</span></a> and are the result of abnormal budding that &#8220;pinches off&#8221; from the tracheal tree in the stage when bronchial buds develop into the primitive of respiratory tree&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">9&#44;10</span></a> Bronchogenic cysts are relatively rare&#44; with a prevalence of 1&#47;68&#44;000&#8226;1&#47;42&#44;000&#46;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">11</span></a> Almost 90&#37; have been reported in the mediastinum&#44; especially in the posterior superior portion&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">12</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Although extremely rare&#44; malignant transformation of a bronchogenic cyst is well known and has been reported in several cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">3&#8226;8</span></a> In a systematic review<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">13</span></a> published in 2010&#44; the authors included 683 adult patients with a diagnosis of bronchogenic cyst&#59; in 5 patients &#40;0&#46;7&#37; of all those studied&#41;&#44; bronchogenic cysts were associated with malignancies&#58; 1 squamous cell&#44; 1 adenocarcinoma&#44; 2 bronchioalveolar carcinomas and 1 large-cell anaplastic carcinoma&#46; It was not stated whether the indication for surgical resection was due to the detection of malignant transformation or whether malignant cells were an incidental finding&#44; but in all cases the carcinoma was found in the cyst wall&#46; To the best of our knowledge&#44; this is the first case of bronchogenic cysts associated with an enteric adenocarcinoma and&#44; in this case&#44; malignant degeneration was the reason for surgical resection&#46; Although there is scant evidence on carcinogenesis in a bronchogenic cyst&#44; some studies suggests that unstable epithelial cells in the cyst wall could have malignant potential and lead to the malignant degeneration&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Surgical excision of bronchogenic cysts has historically been performed for 3 main reasons&#58; &#40;1&#41; to confirm diagnosis&#59; &#40;2&#41; to prevent development of symptoms and&#47;or complications and to pre-empt the possibility of surgery on complex inflammatory lesions&#59; and &#40;3&#41; to avoid any potential for malignant degeneration&#46; However&#44; in the reported case&#44; no surgical treatment was requested until the malignant transformation was discovered 12 years later&#46; Nowadays&#44; the evidence for conservative management of asymptomatic bronchogenic cyst is very limited&#44; and we must take into consideration that approximately 45&#37; of patients who are asymptomatic at the moment of diagnosis will eventually develop symptoms or complications&#44;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">13</span></a> and there is a risk of secondary tumours arising from a bronchogenic cyst&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion&#44; we report the first case of a patient with an enteric adenocarcinoma arising from a longstanding bronchogenic cyst&#46; We consider that surgical excision of bronchogenic cysts should be recommended even in asymptomatic patients&#44; the aim being to preclude malignant degeneration&#44; prevent complications or the development of symptoms&#44; and to confirm diagnosis&#46; Conservative management should be offered only if close long-term follow-up can be guaranteed&#46;</p></span>"
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Scientific Letter
Enteric Adenocarcinoma Arising From a Bronchogenic Cyst
Adenocarcinoma de tipo entèc)rico originado a partir de un quiste broncogèc)nico
Mª Teresa Gómez-Hernández
Corresponding author
, Nuria Novoa, José Luis Aranda, Marcelo F. Jiménez-López
Departamento de Cirugía Torácica, Hospital Universitario de Salamanca, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Bronchogenic cysts are uncommon congenital lesions usually located in the mediastinum or lung and resulting from abnormal budding of foregut in the embryonic stage&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">1&#44;2</span></a> Malignant transformation is very rare and only few well-documented cases have been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">3&#8226;8</span></a> In these cases&#44; histological findings were bronchioalveolar carcinoma&#44; adenocarcinoma&#44; squamous cell carcinoma and large cell carcinoma&#46; To the best of our knowledge&#44; this is the first case of enteric adenocarcinoma arising from an intrapulmonary bronchogenic cyst&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 76-year-old woman with a medical history of hypertension&#44; hypercholesterolemia&#44; hypoacusis&#44; glaucoma&#44; rheumatic polymyalgia and bronchial asthma was referred to our centre for surgical resection of a bronchogenic cyst diagnosed 12 years previously in another hospital&#46; The patient&#44; a farmer&#44; did not have any allergies or toxic habits&#46; She was completely asymptomatic&#44; and physical examination only revealed an absence of breath sounds in the posterior right lower hemithorax&#46; All laboratory tests were normal&#46; The computed tomography &#40;CT&#41; showed a 10&#46;6<span class="elsevierStyleHsp" style=""></span>cm mass in the right lower lobe with calcified walls&#44; which was compatible with bronchogenic cyst &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The bronchoscopy showed an irregular white endobronchial mass at the entrance of the right lower bronchus&#44; which also affected and partially occluded the middle lobe bronchus&#46; The histology of the endobronchial lesion was mucinous adenocarcinoma of the lung&#46; Based on these findings&#44; a PET-CT was performed&#44; which showed an ametabolic mass with a mild increased uptake in the peripheral area and in mediastinal lymph nodes &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The patient underwent functional pulmonary testing and was considered eligible for surgery&#46; She underwent right lower lobectomy with lymph node dissection through muscle-sparing thoracotomy&#46; The postoperative period was uneventful and the patient was discharged 6 days after surgery&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Definitive histology of the specimen was infiltrating colloid adenocarcinoma with immunophenotype of enteric adenocarcinoma arising from a benign cystic mucoid lesion compatible with bronchogenic cyst&#46; Immunohistochemical staining was positive for cytokinin 20 and CDX 2&#44; and negative for cytokinin 20 and TTF1&#46; Mediastinal lymph nodes were not affected&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The final diagnosis was enteric adenocarcinoma arising from a bronchogenic cyst&#46; No additional lesions were found on colonoscopy and further abdominal exams&#46; Because no signs of disease dissemination were found&#44; no additional therapy was applied&#46; The patient is alive without recurrence of disease 6 months after the surgery&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Bronchogenic cysts are congenital lesions thought to originate from the primitive ventral foregut&#44;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">2</span></a> and are the result of abnormal budding that &#8220;pinches off&#8221; from the tracheal tree in the stage when bronchial buds develop into the primitive of respiratory tree&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">9&#44;10</span></a> Bronchogenic cysts are relatively rare&#44; with a prevalence of 1&#47;68&#44;000&#8226;1&#47;42&#44;000&#46;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">11</span></a> Almost 90&#37; have been reported in the mediastinum&#44; especially in the posterior superior portion&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">12</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Although extremely rare&#44; malignant transformation of a bronchogenic cyst is well known and has been reported in several cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">3&#8226;8</span></a> In a systematic review<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">13</span></a> published in 2010&#44; the authors included 683 adult patients with a diagnosis of bronchogenic cyst&#59; in 5 patients &#40;0&#46;7&#37; of all those studied&#41;&#44; bronchogenic cysts were associated with malignancies&#58; 1 squamous cell&#44; 1 adenocarcinoma&#44; 2 bronchioalveolar carcinomas and 1 large-cell anaplastic carcinoma&#46; It was not stated whether the indication for surgical resection was due to the detection of malignant transformation or whether malignant cells were an incidental finding&#44; but in all cases the carcinoma was found in the cyst wall&#46; To the best of our knowledge&#44; this is the first case of bronchogenic cysts associated with an enteric adenocarcinoma and&#44; in this case&#44; malignant degeneration was the reason for surgical resection&#46; Although there is scant evidence on carcinogenesis in a bronchogenic cyst&#44; some studies suggests that unstable epithelial cells in the cyst wall could have malignant potential and lead to the malignant degeneration&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Surgical excision of bronchogenic cysts has historically been performed for 3 main reasons&#58; &#40;1&#41; to confirm diagnosis&#59; &#40;2&#41; to prevent development of symptoms and&#47;or complications and to pre-empt the possibility of surgery on complex inflammatory lesions&#59; and &#40;3&#41; to avoid any potential for malignant degeneration&#46; However&#44; in the reported case&#44; no surgical treatment was requested until the malignant transformation was discovered 12 years later&#46; Nowadays&#44; the evidence for conservative management of asymptomatic bronchogenic cyst is very limited&#44; and we must take into consideration that approximately 45&#37; of patients who are asymptomatic at the moment of diagnosis will eventually develop symptoms or complications&#44;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">13</span></a> and there is a risk of secondary tumours arising from a bronchogenic cyst&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion&#44; we report the first case of a patient with an enteric adenocarcinoma arising from a longstanding bronchogenic cyst&#46; We consider that surgical excision of bronchogenic cysts should be recommended even in asymptomatic patients&#44; the aim being to preclude malignant degeneration&#44; prevent complications or the development of symptoms&#44; and to confirm diagnosis&#46; Conservative management should be offered only if close long-term follow-up can be guaranteed&#46;</p></span>"
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ISSN: 15792129
Original language: English
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