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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Systemic arterialization of the lung without pulmonary sequestration is a rare congenital condition characterized by the presence of an aberrant arterial branch originated from the aorta&#46; This artery supplies a lung with normal parenchyma and bronchial anatomy&#46; Clinically&#44; haemoptysis represents the main clinical manifestation&#46; Few cases have been reported so far and therapeutic management is not standardized&#46; Embolization is a non-invasive treatment with increasing indications&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A thirty-year-old woman was referred to our centre after two episodes of haemoptysis&#46; She was allergic to penicillin with unremarkable comorbidities&#46; No prior treatment except oral contraception&#46; No other bleedings or haemostatic dyscrasias were documented&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The first episode happened one year prior with limited symptoms after conservative treatment in another institution&#46; Chest X-ray&#44; blood and functional lung tests were unremarkable&#46; Eventually&#44; a thoracic computed tomography &#40;CT&#41; angiography showed an anomalous systemic artery arising from the abdominal aorta &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The artery irrigated the posterior-basal segment of right lower lobe &#40;RLL&#41; with no other alterations in pulmonary parenchyma&#46; Pulmonary and cardiac shunts were ruled out&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Treatment by endovascular approach was decided after a multidisciplinary approach with pulmonologists&#44; thoracic and vascular surgeons&#44; and interventional radiologists&#46; The arteriography confirmed the previous findings and normal venous drainage&#46; Finally&#44; endovascular embolization of the anomalous artery was performed using coils and Glubran&#174; with no immediate complications&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Soon after the procedure&#44; the patient presented fever and pleuritic pain due to a mild right pleural effusion and a small area of pulmonary infarct&#44; managed with conventional analgesia&#46; A 3-month-follow-up CT-angiography &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B and C&#41; showed the coils in the anomalous occluded artery with no other remarkable findings&#46; The patient currently remains asymptomatic&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Pulmonary sequestration represents a mass of abnormal&#44; not functional pulmonary tissue supplied by an anomalous systemic artery&#46; Its main feature is its independence from the tracheobronchial tree&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> The term &#8220;sequestration&#8221; was introduced by Pryce<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">2</span></a> to describe congenital abnormalities characterized by anomalous systemic arterial supply to the lung&#44; related with atresia or hypoplasia of the pulmonary artery&#46; Since then&#44; the spectrum of bronchopulmonary vascular malformations has grown widely&#44; especially with the &#8220;sequestration spectrum&#8221; concept&#44; in order to include malformations that do not fulfil the original sequestration definition&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> Additionally&#44; the term &#8220;pulmonary malinosculation&#8221; gathered all congenital lung abnormalities with anomalous communication between blood vessels or other tubular structures&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The presence of normal lung parenchyma and bronchial supply is the main difference between systemic arterialization of the lung without pulmonary sequestration and true sequestration&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">4</span></a> The former is rare&#44; with few cases reported&#44;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">4&#8211;6</span></a> and consists of an aberrant systemic arterial branch arising from the thoracic descending aorta or the abdominal or celiac axis&#46; Usually&#44; the artery coming from the abdominal aorta supplies the RLL&#44; whereas the thoracic origin often supplies the left lower lobe&#44;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> the most commonly involved segment&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In our case&#44; the anomalous artery supplied a non-sequestered region in the RLL without other congenital abnormalities&#46; The aetiology of this condition is unknown&#46; Persistence of an embryonic connection between the aorta and the pulmonary parenchyma remains the main hypothesis&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">8</span></a> Most patients are asymptomatic&#44;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">9</span></a> although recurrent pulmonary infection and haemoptysis are possible clinical manifestations&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> One explanation for the intermittent haemoptysis&#44; present in our case&#44; is the alveolar haemorrhage secondary to the high vascular pressure in the abnormally perfused segments&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The treatment is generally recommended even in asymptomatic patients in order to prevent possible fatal haemoptysis&#46; The therapeutic approach is not fully standardized although the most widespread consensual treatment in sequestration currently is surgical resection&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">10</span></a> Surgical approach includes lobectomy&#44; systemic artery-to-pulmonary artery anastomosis and endovascular occlusion of the aberrant systemic artery&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">11</span></a> Less invasive approach with embolization has also been described in few cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0105"><span class="elsevierStyleSup">6&#44;11&#44;12</span></a> Multiple substances may be used to perform embolization&#46; We decided to use an embolic liquid agent combined with coils&#46; The embolic liquid agent reduces the risk of rebleeding<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">13&#44;14</span></a> although distal embolization and tissue necrosis might be higher&#46;<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">15</span></a> Coils allow proximal occlusion and collateral flow&#46; In our case&#44; the calibre of the artery and the large irrigated territory may justify the lung infarction independently of the Glubran use&#46; The patient presented a mild post-embolization syndrome &#40;PES&#41;&#44; the most common side effect of embolization&#46; PES is characterized by fever&#44; nausea and pain&#44; usually within the first 72<span class="elsevierStyleHsp" style=""></span>h after the procedure&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; our case emphasizes the fact that the embolization is a safe and feasible alternative to surgery&#44; proving to be a procedure with few complications and clinically successful&#46; Further studies are now warranted to study the benefit over surgery in patients with anomalous vascularization of the lung&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical statement</span><p id="par0055" class="elsevierStylePara elsevierViewall">This work was conducted at Hospital de la Santa Creu i Sant Pau with approval from the ethic committee of our Hospital&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">No funding was received for this research&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare no conflicts of interest&#46;</p></span></span>"
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Scientific letter
Recurrent Haemoptysis Secondary to Abnormal Arterial Supply to the Right Lower Lobe of the Lung
Hemoptisis recurrente secundaria a vascularización anómala del lóbulo inferior del pulmón derecho
Paloma Millán Billia,b,
Corresponding author
pmillanbilli@gmail.com

Corresponding author.
, Virginia Pajares Ruizb, Rubén Guerrero Varac, Jordi Villalba Auñónc, Alfons Torrego Fernandezb
a Department of Respiratory Diseases, Hospital Universitari Germans Trias i Pujol, Badalona, Spain
b Department of Respiratory Diseases, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain
c Department of Interventional Radiology, Hospital de la Santa Creu I Sant Pau, Barcelona, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Systemic arterialization of the lung without pulmonary sequestration is a rare congenital condition characterized by the presence of an aberrant arterial branch originated from the aorta&#46; This artery supplies a lung with normal parenchyma and bronchial anatomy&#46; Clinically&#44; haemoptysis represents the main clinical manifestation&#46; Few cases have been reported so far and therapeutic management is not standardized&#46; Embolization is a non-invasive treatment with increasing indications&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A thirty-year-old woman was referred to our centre after two episodes of haemoptysis&#46; She was allergic to penicillin with unremarkable comorbidities&#46; No prior treatment except oral contraception&#46; No other bleedings or haemostatic dyscrasias were documented&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The first episode happened one year prior with limited symptoms after conservative treatment in another institution&#46; Chest X-ray&#44; blood and functional lung tests were unremarkable&#46; Eventually&#44; a thoracic computed tomography &#40;CT&#41; angiography showed an anomalous systemic artery arising from the abdominal aorta &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The artery irrigated the posterior-basal segment of right lower lobe &#40;RLL&#41; with no other alterations in pulmonary parenchyma&#46; Pulmonary and cardiac shunts were ruled out&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Treatment by endovascular approach was decided after a multidisciplinary approach with pulmonologists&#44; thoracic and vascular surgeons&#44; and interventional radiologists&#46; The arteriography confirmed the previous findings and normal venous drainage&#46; Finally&#44; endovascular embolization of the anomalous artery was performed using coils and Glubran&#174; with no immediate complications&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Soon after the procedure&#44; the patient presented fever and pleuritic pain due to a mild right pleural effusion and a small area of pulmonary infarct&#44; managed with conventional analgesia&#46; A 3-month-follow-up CT-angiography &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B and C&#41; showed the coils in the anomalous occluded artery with no other remarkable findings&#46; The patient currently remains asymptomatic&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Pulmonary sequestration represents a mass of abnormal&#44; not functional pulmonary tissue supplied by an anomalous systemic artery&#46; Its main feature is its independence from the tracheobronchial tree&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> The term &#8220;sequestration&#8221; was introduced by Pryce<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">2</span></a> to describe congenital abnormalities characterized by anomalous systemic arterial supply to the lung&#44; related with atresia or hypoplasia of the pulmonary artery&#46; Since then&#44; the spectrum of bronchopulmonary vascular malformations has grown widely&#44; especially with the &#8220;sequestration spectrum&#8221; concept&#44; in order to include malformations that do not fulfil the original sequestration definition&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> Additionally&#44; the term &#8220;pulmonary malinosculation&#8221; gathered all congenital lung abnormalities with anomalous communication between blood vessels or other tubular structures&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The presence of normal lung parenchyma and bronchial supply is the main difference between systemic arterialization of the lung without pulmonary sequestration and true sequestration&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">4</span></a> The former is rare&#44; with few cases reported&#44;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">4&#8211;6</span></a> and consists of an aberrant systemic arterial branch arising from the thoracic descending aorta or the abdominal or celiac axis&#46; Usually&#44; the artery coming from the abdominal aorta supplies the RLL&#44; whereas the thoracic origin often supplies the left lower lobe&#44;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> the most commonly involved segment&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In our case&#44; the anomalous artery supplied a non-sequestered region in the RLL without other congenital abnormalities&#46; The aetiology of this condition is unknown&#46; Persistence of an embryonic connection between the aorta and the pulmonary parenchyma remains the main hypothesis&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">8</span></a> Most patients are asymptomatic&#44;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">9</span></a> although recurrent pulmonary infection and haemoptysis are possible clinical manifestations&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> One explanation for the intermittent haemoptysis&#44; present in our case&#44; is the alveolar haemorrhage secondary to the high vascular pressure in the abnormally perfused segments&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The treatment is generally recommended even in asymptomatic patients in order to prevent possible fatal haemoptysis&#46; The therapeutic approach is not fully standardized although the most widespread consensual treatment in sequestration currently is surgical resection&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">10</span></a> Surgical approach includes lobectomy&#44; systemic artery-to-pulmonary artery anastomosis and endovascular occlusion of the aberrant systemic artery&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">11</span></a> Less invasive approach with embolization has also been described in few cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0105"><span class="elsevierStyleSup">6&#44;11&#44;12</span></a> Multiple substances may be used to perform embolization&#46; We decided to use an embolic liquid agent combined with coils&#46; The embolic liquid agent reduces the risk of rebleeding<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">13&#44;14</span></a> although distal embolization and tissue necrosis might be higher&#46;<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">15</span></a> Coils allow proximal occlusion and collateral flow&#46; In our case&#44; the calibre of the artery and the large irrigated territory may justify the lung infarction independently of the Glubran use&#46; The patient presented a mild post-embolization syndrome &#40;PES&#41;&#44; the most common side effect of embolization&#46; PES is characterized by fever&#44; nausea and pain&#44; usually within the first 72<span class="elsevierStyleHsp" style=""></span>h after the procedure&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; our case emphasizes the fact that the embolization is a safe and feasible alternative to surgery&#44; proving to be a procedure with few complications and clinically successful&#46; Further studies are now warranted to study the benefit over surgery in patients with anomalous vascularization of the lung&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical statement</span><p id="par0055" class="elsevierStylePara elsevierViewall">This work was conducted at Hospital de la Santa Creu i Sant Pau with approval from the ethic committee of our Hospital&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">No funding was received for this research&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare no conflicts of interest&#46;</p></span></span>"
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Article information
ISSN: 03002896
Original language: English
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