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the case was presented before the multidisciplinary committee&#44; and the decision was taken to start antiparasitic treatment with albendazole &#40;400<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h&#41; and praziquantel &#40;1800<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h&#41;&#44; with follow-up CT for evaluating treatment response and subsequent surgical resection&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">After 6 weeks of antiparasitic treatment&#44; the radiological findings showed little reduction in mass size and persisting internal cavitated areas&#44; along with significant neovascularization originating in the thoracic artery via the bronchial arteries&#44; and in the celiac trunk&#44; via the arteries of the diaphragm&#46; The patient&#39;s lung function test results were within normal limits&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Surgical resection was performed&#44; 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and referred to the infectious diseases department for follow-up and control of his medical treatment&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Hydatidosis is a parasitic infection caused by the larval forms of <span class="elsevierStyleItalic">Echinococcus granulosus</span>&#46; The most commonly affected organs are the liver &#40;70&#37;&#41; and the lungs &#40;25&#37;&#41;&#46; About 10&#37;&#8211;15&#37; of the embryos can spread to other organs via the systemic circulation&#44; including the chambers of the heart&#44; interventricular septum and pericardium &#40;0&#46;02&#37;&#8211;2&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> The pulmonary arteries are an exceptionally rare site of involvement&#46; In our case&#44; the hydatid cyst was located at the level of the right lower lobe&#44; within the right lower lobe artery&#44; presumably due to the parasite implanting in the arterial lumen after crossing the wall through small lesions in the intima&#44; aneurysms&#44; or via the feeder vessels&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">A hydatid cyst located within the pulmonary artery is associated with high mortality rates due to secondary dissemination&#44; embolization&#44; or anaphylactic shock after rupture&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> Hydatid cysts are known to grow 2&#8211;3<span class="elsevierStyleHsp" style=""></span>cm&#47;year&#46; Within vessels&#44; cysts grow slowly inside the lumen until it is obstructed&#46; In pulmonary arteries&#44; this process may be sufficiently slow to allow pulmonary perfusion to be adequately established via the bronchial arteries or other collateral branches&#46; This explains why patients may be initially asymptomatic&#44; and why our patient had no signs of occlusion despite total occlusion of the right lower lobe artery&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The differential diagnosis of hydatid cysts in the pulmonary artery should include pulmonary thromboembolism &#40;fat&#44; amniotic fluid&#44; septic emboli&#44; exogenous material&#41; and primary tumors of the pulmonary artery&#44; such as sarcomas&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Due to the high risk of cyst rupture and anaphylactic shock&#44; surgery should be performed as soon as possible after diagnosis&#46; The extraction of endovascular cysts even in asymptomatic patients is recommended&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> As this form of hydatidosis is so rare&#44; no standardized surgical techniques have been described&#44; so treatment must be individualized&#46; The approach will depend on the adherence of the hydatid cyst to the arterial wall and its anatomical location&#46; Surgical treatment must focus on preventing cyst rupture and migration&#44; which may have fatal consequences&#44; such as secondary dissemination&#44; embolism&#44; anaphylactic shock&#44; and the formation of pseudoaneurysms&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> In our case&#44; we clamped the right pulmonary artery to avoid such complications&#46; Unilateral obstruction of the pulmonary artery can be successfully treated with arteriotomy and cyst resection with or without lung resection&#44; or with pulmonary endarterectomy&#46; In situations of concomitant pulmonary and arterial involvement&#44; a 2-stage surgical intervention can be considered&#58; first&#44; resection of the intra-arterial hydatid cyst&#44; followed by cystectomy or lung resection&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Recommended treatment of intra-arterial hydatidosis is surgical intervention followed by complementary medical treatment&#46; CT follow-up is indicated to detect possible recurrences or development of pseudoaneurysms&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p></span>"
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Scientific Letter
An Unusual Occupant of the Pulmonary Artery
Un ocupante insólito de la arteria pulmonar
M. Teresa Gómez Hernándeza,
Corresponding author
, María Rodríguez Péreza, Pilar García Hernándezb, Marcelo F. Jiménez Lópeza
a Departamento de Cirugía Torácica, Hospital Universitario de Salamanca, Salamanca, Spain
b Servicio de Radiodiagnóstico, Hospital Universitario de Salamanca, Salamanca, Spain
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The chest radiograph revealed a polylobulated mass in the right lower lobe with hilar thickening&#44; initially suggestive of lung cancer&#46; Chest computed tomography showed a mass measuring 11<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>8&#46;8<span class="elsevierStyleHsp" style=""></span>cm in the right lower lobe with hypodense areas suggestive of cysts&#46; The mass showed a 10<span class="elsevierStyleHsp" style=""></span>cm contact with pleural surface&#44; extending with peribronchovascular distribution toward the hilar region&#46; Multiple nodular images of similar characteristics measuring up to 4&#46;5<span class="elsevierStyleHsp" style=""></span>cm were seen inside the artery&#46; Radiological findings were suggestive of pulmonary hydatidosis with invasion of the right lower lobe artery &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#8211;C&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">After serological confirmation of <span class="elsevierStyleItalic">Echinococcus granulosus</span> infection&#44; the case was presented before the multidisciplinary committee&#44; and the decision was taken to start antiparasitic treatment with albendazole &#40;400<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h&#41; and praziquantel &#40;1800<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h&#41;&#44; with follow-up CT for evaluating treatment response and subsequent surgical resection&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">After 6 weeks of antiparasitic treatment&#44; the radiological findings showed little reduction in mass size and persisting internal cavitated areas&#44; along with significant neovascularization originating in the thoracic artery via the bronchial arteries&#44; and in the celiac trunk&#44; via the arteries of the diaphragm&#46; The patient&#39;s lung function test results were within normal limits&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Surgical resection was performed&#44; and antiparasitic treatment continued throughout the peri-operative period&#46; Intraoperative findings included hepatization of the lower right lobe&#44; significant vascularization originating in the bronchial arteries&#44; and increased caliber of the right lower lobe artery&#46; After control and clamping of the right pulmonary artery at the extrapericardiac origin&#44; arteriotomy was performed at the lower lobe branch&#44; and the intravascular hydatid cysts were extracted &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D&#41;&#46; The proximal lumen of the pulmonary artery was aspirated to the level of the clamp&#46; Given the significant perihilar inflammatory reaction&#44; the proximal intermediate bronchus could not be resected&#44; so a middle-lower bilobectomy was performed&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The only post-operative complication presented by the patient was prolonged air leak&#46; He was discharged on day 8 after surgery&#44; and referred to the infectious diseases department for follow-up and control of his medical treatment&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Hydatidosis is a parasitic infection caused by the larval forms of <span class="elsevierStyleItalic">Echinococcus granulosus</span>&#46; The most commonly affected organs are the liver &#40;70&#37;&#41; and the lungs &#40;25&#37;&#41;&#46; About 10&#37;&#8211;15&#37; of the embryos can spread to other organs via the systemic circulation&#44; including the chambers of the heart&#44; interventricular septum and pericardium &#40;0&#46;02&#37;&#8211;2&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> The pulmonary arteries are an exceptionally rare site of involvement&#46; In our case&#44; the hydatid cyst was located at the level of the right lower lobe&#44; within the right lower lobe artery&#44; presumably due to the parasite implanting in the arterial lumen after crossing the wall through small lesions in the intima&#44; aneurysms&#44; or via the feeder vessels&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">A hydatid cyst located within the pulmonary artery is associated with high mortality rates due to secondary dissemination&#44; embolization&#44; or anaphylactic shock after rupture&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> Hydatid cysts are known to grow 2&#8211;3<span class="elsevierStyleHsp" style=""></span>cm&#47;year&#46; Within vessels&#44; cysts grow slowly inside the lumen until it is obstructed&#46; In pulmonary arteries&#44; this process may be sufficiently slow to allow pulmonary perfusion to be adequately established via the bronchial arteries or other collateral branches&#46; This explains why patients may be initially asymptomatic&#44; and why our patient had no signs of occlusion despite total occlusion of the right lower lobe artery&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The differential diagnosis of hydatid cysts in the pulmonary artery should include pulmonary thromboembolism &#40;fat&#44; amniotic fluid&#44; septic emboli&#44; exogenous material&#41; and primary tumors of the pulmonary artery&#44; such as sarcomas&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Due to the high risk of cyst rupture and anaphylactic shock&#44; surgery should be performed as soon as possible after diagnosis&#46; The extraction of endovascular cysts even in asymptomatic patients is recommended&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> As this form of hydatidosis is so rare&#44; no standardized surgical techniques have been described&#44; so treatment must be individualized&#46; The approach will depend on the adherence of the hydatid cyst to the arterial wall and its anatomical location&#46; Surgical treatment must focus on preventing cyst rupture and migration&#44; which may have fatal consequences&#44; such as secondary dissemination&#44; embolism&#44; anaphylactic shock&#44; and the formation of pseudoaneurysms&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> In our case&#44; we clamped the right pulmonary artery to avoid such complications&#46; Unilateral obstruction of the pulmonary artery can be successfully treated with arteriotomy and cyst resection with or without lung resection&#44; or with pulmonary endarterectomy&#46; In situations of concomitant pulmonary and arterial involvement&#44; a 2-stage surgical intervention can be considered&#58; first&#44; resection of the intra-arterial hydatid cyst&#44; followed by cystectomy or lung resection&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Recommended treatment of intra-arterial hydatidosis is surgical intervention followed by complementary medical treatment&#46; CT follow-up is indicated to detect possible recurrences or development of pseudoaneurysms&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p></span>"
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Article information
ISSN: 15792129
Original language: English
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