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hysterectomy&#44; and double adnexectomy&#46; Physical examination was unremarkable&#44; and both complete blood count and biochemistry results were within normal ranges&#46; Chest X-ray showed the presence of a round subcarinal lesion&#46; CT confirmed the presence of a mass enhanced by administration of contrast medium&#44; measuring about 8&#46;5&#215;4&#46;5<span class="elsevierStyleHsp" style=""></span>cm in diameter&#44; located immediately behind the right pulmonary artery and accompanied by paraaortic and retroperitoneal lymphadenopathies&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Fiberoptic bronchoscopy &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41; showed a purplish mass with a polylobulated surface located in the main carina&#44; almost entirely occluding the entrance to the left main bronchus&#44; but that did not prevent passage of the bronchoscope&#46; The appearance of the endobronchial lesion and the patient&#39;s history of an intrathoracic vascular lesion made the collection of endoscopic biopsies inadvisable&#46; Some days later&#44; rigid bronchoscopy was performed under general anesthesia&#44; and biopsies were collected&#44; complicated by severe bleeding controlled with diathermy&#44; followed by arteriography with embolization of the branches feeding the right bronchial artery&#46; Biopsy samples showed the presence of numerous endothelized vessels with fibrous walls and a dense proliferation of cells with elongated nuclei arranged in bundles with variable spatial arrangement with no necrosis or mitosis&#44; that were positive for markers CD34 and CD31&#44; Ki-67 in less than 5&#37;&#44; and negative for epithelial and muscle markers&#59; these findings were consistent with pulmonary hemangiopericytoma&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Between 2&#37; and 6&#37; of all primary mediastinal tumors are of mesenchymal origin&#46; Of these&#44; hemangiopericytoma is a rarity&#58; it originates in the Zimmermann pericytes&#44; which form part of the outer layer surrounding the endothelium of the capillaries and is now classified as perivascular tumor&#46; It represents less than 2&#37; of all soft tissue sarcomas&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a> and approximately 1&#37; of all tumors of vascular origin&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> The main sites tend to be the muscle tissue of the limbs&#44; subcutaneous tissue&#44; and the retroperitoneum&#46; The chest &#40;usually mediastinal&#41; as the primary site of hemangiopericytoma is extremely rare&#44; as attested by a review of literature which retrieved very few cases&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> Published cases of pulmonary involvement mostly began as solitary pulmonary nodules&#44; and to our knowledge&#44; this is the first case with endobronchial expression evidenced by bronchoscopy&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">This type of tumor usually begins with a wide variety of symptoms&#44; which most notably include hemoptysis&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> Image testing&#44; especially CT with and without contrast and magnetic resonance imaging &#40;MRI&#41;&#44;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1&#44;2</span></a> helps to visualize the lesion and point to its vascular origin&#44; although these tests are not diagnostic in themselves&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Pericytomas tend to behave unpredictably&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">5&#44;6</span></a> In the case of our patient&#44; after the initial diagnosis of angioma&#44; the lesion grew slowly without showing any clinical signs or symptoms that would suggest malignancy&#46; Findings that suggest a more aggressive behavior are&#58; size greater than 10<span class="elsevierStyleHsp" style=""></span>cm &#40;with a 66&#37; probability of metastasis&#41;&#44; the existence of more than 3 mitoses per field&#44; necrosis&#44; pleural invasion&#44; and vascular invasion&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">7</span></a> In our case&#44; despite the size of the lesion &#40;8&#46;5&#215;4&#46;5<span class="elsevierStyleHsp" style=""></span>cm&#41;&#44; the absence of necrosis or mitosis on the pathology study suggested a lack of aggressive behavior up to the time of diagnosis&#46; It is clear however that&#44; since it was a large central endobronchial vascular tumor&#44; the risk of potentially fatal local complications was high&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Treatment of tumors of this type is based primarily on surgical resection of the lesion&#44; if possible&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a> Prior embolization of the feeder arterial branches is always advisable&#46; 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Scientific Letter
Primary Pulmonary Hemangiopericytoma
Hemangiopericitoma pulmonar primario
José María Hernández Péreza,
Corresponding author
jmherper@hotmail.com

Corresponding author.
, Lorenzo Pérez Negrínb, Claudia Viviana López Charryb
a Sección de Neumología, Hospital General de La Palma, Breña Alta, La Palma, Santa Cruz de Tenerife, Spain
b Servicio de Neumología, Hospital Universitario Nuestra Señora de Candelaria, Santa Cruz de Tenerife, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The high prevalence of epithelial lung tumors means that mesenchymal lung tumors are rarities seen very occasionally in routine clinical practice&#46; We report the case of a patient referred to the respiratory medicine clinic for the study of recurrent self-limiting episodes of bloody expectoration&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">This was a female patient&#44; 56 years of age&#44; originally from Venezuela&#44; who presented due to repeated self-limiting episodes of expectoration of blood while coughing&#44; not affecting hemodynamic parameters&#44; blood gases&#44; or blood counts&#46; Four years previously in her home country&#44; she had been diagnosed on CT with a prevertebral solid tumor&#44; determined on thoracotomy to be thoracic hemangioma&#44; that was treated by radiation therapy &#40;total dose unknown&#41;&#46; Other surgical history included left inguinal herniorrhaphy&#44; hysterectomy&#44; and double adnexectomy&#46; Physical examination was unremarkable&#44; and both complete blood count and biochemistry results were within normal ranges&#46; Chest X-ray showed the presence of a round subcarinal lesion&#46; CT confirmed the presence of a mass enhanced by administration of contrast medium&#44; measuring about 8&#46;5&#215;4&#46;5<span class="elsevierStyleHsp" style=""></span>cm in diameter&#44; located immediately behind the right pulmonary artery and accompanied by paraaortic and retroperitoneal lymphadenopathies&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Fiberoptic bronchoscopy &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41; showed a purplish mass with a polylobulated surface located in the main carina&#44; almost entirely occluding the entrance to the left main bronchus&#44; but that did not prevent passage of the bronchoscope&#46; The appearance of the endobronchial lesion and the patient&#39;s history of an intrathoracic vascular lesion made the collection of endoscopic biopsies inadvisable&#46; Some days later&#44; rigid bronchoscopy was performed under general anesthesia&#44; and biopsies were collected&#44; complicated by severe bleeding controlled with diathermy&#44; followed by arteriography with embolization of the branches feeding the right bronchial artery&#46; Biopsy samples showed the presence of numerous endothelized vessels with fibrous walls and a dense proliferation of cells with elongated nuclei arranged in bundles with variable spatial arrangement with no necrosis or mitosis&#44; that were positive for markers CD34 and CD31&#44; Ki-67 in less than 5&#37;&#44; and negative for epithelial and muscle markers&#59; these findings were consistent with pulmonary hemangiopericytoma&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Between 2&#37; and 6&#37; of all primary mediastinal tumors are of mesenchymal origin&#46; Of these&#44; hemangiopericytoma is a rarity&#58; it originates in the Zimmermann pericytes&#44; which form part of the outer layer surrounding the endothelium of the capillaries and is now classified as perivascular tumor&#46; It represents less than 2&#37; of all soft tissue sarcomas&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a> and approximately 1&#37; of all tumors of vascular origin&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> The main sites tend to be the muscle tissue of the limbs&#44; subcutaneous tissue&#44; and the retroperitoneum&#46; The chest &#40;usually mediastinal&#41; as the primary site of hemangiopericytoma is extremely rare&#44; as attested by a review of literature which retrieved very few cases&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> Published cases of pulmonary involvement mostly began as solitary pulmonary nodules&#44; and to our knowledge&#44; this is the first case with endobronchial expression evidenced by bronchoscopy&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">This type of tumor usually begins with a wide variety of symptoms&#44; which most notably include hemoptysis&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> Image testing&#44; especially CT with and without contrast and magnetic resonance imaging &#40;MRI&#41;&#44;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1&#44;2</span></a> helps to visualize the lesion and point to its vascular origin&#44; although these tests are not diagnostic in themselves&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Pericytomas tend to behave unpredictably&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">5&#44;6</span></a> In the case of our patient&#44; after the initial diagnosis of angioma&#44; the lesion grew slowly without showing any clinical signs or symptoms that would suggest malignancy&#46; Findings that suggest a more aggressive behavior are&#58; size greater than 10<span class="elsevierStyleHsp" style=""></span>cm &#40;with a 66&#37; probability of metastasis&#41;&#44; the existence of more than 3 mitoses per field&#44; necrosis&#44; pleural invasion&#44; and vascular invasion&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">7</span></a> In our case&#44; despite the size of the lesion &#40;8&#46;5&#215;4&#46;5<span class="elsevierStyleHsp" style=""></span>cm&#41;&#44; the absence of necrosis or mitosis on the pathology study suggested a lack of aggressive behavior up to the time of diagnosis&#46; It is clear however that&#44; since it was a large central endobronchial vascular tumor&#44; the risk of potentially fatal local complications was high&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Treatment of tumors of this type is based primarily on surgical resection of the lesion&#44; if possible&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a> Prior embolization of the feeder arterial branches is always advisable&#46; Postoperative radiation therapy also plays an important role in lesions of this type&#44; while chemotherapy appears to have no clear benefit and is reserved for selected cases and always administered with palliative intent&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In our case&#44; the site of the lesion and the treatments received earlier ruled out surgical resection&#44; so we opted for tumor embolization&#44; implantation of a silicone Dumon Y stent&#44; and external radiation therapy&#46; This prosthesis had to be removed 6 months later due to intractable cough&#44; after which successive bronchial dilation procedures were performed using rigid bronchoscopy&#44; during the last of which massive bronchial bleeding occurred due to rupture of the tumor mass that resulted in the death of the patient&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Hern&#225;ndez P&#233;rez JM&#44; P&#233;rez Negr&#237;n L&#44; L&#243;pez Charry CV&#46; Hemangiopericitoma pulmonar primario&#46; Arch Bronconeumol&#46; 2019&#59;55&#58;593&#8211;594&#46;</p>"
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Article information
ISSN: 15792129
Original language: English
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