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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Scimitar syndrome &#40;SS&#41; is a rare and complex congenital anomaly&#44; characterized by partial or complete anomalous pulmonary venous drainage from the right or left lung into the inferior vena cava &#40;IVC&#41;&#44; though drainage into the hepatic vein&#44; right atrium &#40;RA&#41; or left atrium &#40;LA&#41;&#44; or the portal vein can also occur&#46; According to the original definition&#44; the syndrome is commonly associated with hypoplasia of the right lung&#44; pulmonary sequestration&#44; persistent left superior vena cava&#44; and dextroposition of the heart&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> In two-thirds of cases&#44; the scimitar vein provides drainage for the entire right lung&#44; but in one third&#44; this vein drains only the lower portion of the right lung&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Partial abnormalities of pulmonary venous return &#40;PAPVR&#41; are seen in 0&#46;4&#8211;0&#46;7&#37; of adult autopsies&#44; while patients with SS account for 3&#8211;5&#37; of these cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a> SS constitutes 0&#46;5&#8211;1&#37; of all congenital heart defects&#46; This rare anomaly has an incidence of approximately 1-3 per 100<span class="elsevierStyleHsp" style=""></span>000 live births&#59; however&#44; the true incidence may be higher because many patients remain asymptomatic&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The etiology is not completely understood&#46; In several patients with total anomalous pulmonary venous return&#44; the gene locus has been mapped to chromosome 4q12&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> and females are more frequently affected than males&#46; The scimitar sign refers to the crescent &#40;resembling the Turkish sword&#41; described by the descent of the anomalous pulmonary vein &#40;the tip of the crescent points inferiorly and medially to the diaphragm&#47;right heart border junction&#41;&#46; The concavity of the crescent is adjacent to the junction of the diaphragm and right heart border&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 44-year-old man presented with several weeks of palpitations&#46; He also described symptoms of dyspnea with mild exertion and substernal chest discomfort at rest&#46; He had a history of long-standing systemic hypertension&#46; The 12-lead ECG showed atrial fibrillation with rapid ventricular rate&#46; His 2D Doppler echocardiogram revealed a mildly enlarged RA and right ventricle &#40;RV&#41;&#46; He subsequently underwent right and left heart catheterization with selective coronary angiography&#46; Right and left heart catheterization with a full oximetry run to calculate shunts revealed Qp&#47;Qs of 1&#46;3&#46; Selective coronary angiography was performed using right radial artery access&#44; and showed no significant coronary disease&#46; However&#44; angiography of the aortic arch showed a single trunk takeoff for the large vessels from the aortic arch &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41;&#46; Using right femoral vein access&#44; a 5-Fr multipurpose diagnostic catheter was advanced into the upper &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41; and middle &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>&#41; right pulmonary veins as they opened into the superior vena cava &#40;SVC&#41;&#46; To exclude any possible associated atrial septal defect&#44; an MP-1 catheter was engaged into what proved to be a coronary-cameral fistula &#40;CCF&#41; opening into the RA separately from the coronary sinus &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Figure 4</a>&#41;&#44; the CCF went from the coronary vein to the RA and the coronary sinus was also filled with contrast retrogradely from the vein &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Figure 4</a>&#41;&#46; Pulmonary angiography using a 5-Fr pigtail catheter showed a moderately dilated pulmonary trunk &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Figure 5</a>&#41;&#46; Following consultations with the cardiothoracic surgery and pediatric cardiology teams&#44; it was felt that the best course of management would be to follow the patient clinically with serial echocardiography&#44; as there was no significant right-to-left shunt&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">SS is a complex form of PAPVR&#44; which is a connection failure between the right pulmonary veins and the LA during fetal development&#46; Variations in PAPVR include the right pulmonary veins draining into the SVC-LA junction&#44; RA&#44; or IVC&#44; or as in our case&#44; separately to a high SVC&#46; In SS&#44; an anomalous right pulmonary vein generally draining the entire right lung but occasionally the middle and superior lobes&#44; may descend in a cephalad-to-caudal direction toward the diaphragm with a crescent &#40;scimitar&#41; shape&#46; This vein then curves sharply to the left just above or below the IVC-RA junction&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> The anomalous right pulmonary venous trunk usually courses anterior to the hilum of the right lung and connects to the IVC just superior to the orifices of the hepatic veins&#46; Rarely&#44; it drains into the left atrium&#44; with normal venous return&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In this patient&#44; the right upper and middle pulmonary veins drained into the high SVC and then ultimately into the RA&#44; leading to increased pulmonary circulation as shown by the enlarged right chambers and main pulmonary artery&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The most common aortic arch branching pattern in humans consists of three great vessels originating from the arch of the aorta&#58; the innominate&#44; left common carotid and left subclavian arteries&#46; In &#8220;bovine&#8221; aortic arch&#44; rather than arising directly from the aortic arch as a separate branch&#44; the left common carotid artery origin is moved to the right and merges with the origin of the innominate artery&#44; or the left common carotid artery originates directly from the innominate artery rather than as a common trunk&#46; A single brachiocephalic trunk originating from the aortic arch and then dividing into right and left brachiocephalic trunks or trifurcating to brachiocephalic&#44; left common carotid and subclavian artery is a very rare anomaly&#44; with few cases reported in the literature&#44;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10&#44;11</span></a> and there is no previously reported case of SS with this anomaly&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Our patient had a venous CCF draining into the RA from the coronary vein in addition to the right upper and middle pulmonary veins draining through the SVC&#46; There is no previously reported case of SS associated with venous CCF in the literature&#46; Coronary-cameral fistulas are unusual congenital or acquired anomalous communications between an epicardial coronary artery and a cardiac chamber&#58; the RA&#44; coronary sinus&#44; right ventricle&#44; left atrium&#44; or left ventricle&#46; In a review of 304 patients with CCF and coronary vascular fistula &#40;CVF&#41;&#44; a continuous cardiac murmur was heard in 82&#37; of the subjects&#46; Dyspnea&#44; chest pain and angina pectoris&#44; palpitations&#44; and fatigue are reported symptoms in clinical presentations&#46; Although rare&#44; infective endocarditis has been reported in patients with CCF&#44; but not in those with CVF&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Surgical repair of SS consists of redirecting the pulmonary venous drainage into the LA&#44; by either baffling the anomalous drainage into the LA via a tunnel or transecting the &#8220;scimitar drainage&#8221; near its entrance into the IVC and then reimplanting it directly into the LA&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> The surgical option is&#44; however&#44; associated with a relatively high incidence of interruption failure&#44; reinserted pulmonary vein stenosis&#44; surgical complications&#44; and redo procedures&#46; Surgical correction is considered in symptomatic patients or in those with increased pulmonary blood flow and signs of right heart dilation with a significant shunt &#40;Qp&#47;Qs &#8805;1&#46;5&#41;&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In the most common form of SS&#44; there is anomalous right pulmonary vein drainage into the IVC with anomalous systemic arterial supply &#40;ASAS&#41; from the abdominal aorta toward the affected pulmonary parenchyma&#44; leading to pulmonary hypertension and subsequent volume overload and heart failure&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">If the presence of a large ASAS to the right lung with pulmonary overcirculation is detected preoperatively &#40;which it was not in our case&#41;&#44; occlusion of the ASAS is frequently performed percutaneously to encourage compensatory growth of the remaining normal lung tissue and to reduce both the associated left-to-right shunt and the risk of chronic and recurrent infection in the abnormal lung in later life&#46; Successful percutaneous occlusion of ASAS in infants with SS has emerged as an acceptable treatment using the Amplatzer&#8482; vascular plug device &#40;AGA Medical&#44; Golden Valley&#44; MN&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13&#44;14</span></a> In a study of 16 infants who underwent percutaneous ASAS interruption&#44; Uthaman et al&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> reported significant reduction in left-to-right shunt and pulmonary artery pressures in &#62;90&#37; of the patients&#44; which translated into symptomatic improvement as well as growth of the right lung in follow-up&#46; Furthermore&#44; this approach leads to sustained long-term clinical improvement without need for further surgical correction in the majority of patients&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; in this case report we describe a patient with scimitar syndrome with very unusual associated anomalies of a single aortic trunk and venous CCF&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Ethical disclosures</span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Protection of human and animal subjects</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Confidentiality of data</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data and that all the patients included in the study received sufficient information and gave their written informed consent to participate in the study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Right to privacy and informed consent</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article&#46; 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        1 => array:2 [
          "identificador" => "xpalclavsec237851"
          "titulo" => "Keywords"
        ]
        2 => array:2 [
          "identificador" => "xres250282"
          "titulo" => "Resumo"
        ]
        3 => array:2 [
          "identificador" => "xpalclavsec237852"
          "titulo" => "Palavras-chave"
        ]
        4 => array:2 [
          "identificador" => "sec0005"
          "titulo" => "Introduction"
        ]
        5 => array:2 [
          "identificador" => "sec0010"
          "titulo" => "Case report"
        ]
        6 => array:2 [
          "identificador" => "sec0015"
          "titulo" => "Discussion"
        ]
        7 => array:3 [
          "identificador" => "sec0020"
          "titulo" => "Ethical disclosures"
          "secciones" => array:3 [
            0 => array:2 [
              "identificador" => "sec0025"
              "titulo" => "Protection of human and animal subjects"
            ]
            1 => array:2 [
              "identificador" => "sec0030"
              "titulo" => "Confidentiality of data"
            ]
            2 => array:2 [
              "identificador" => "sec0035"
              "titulo" => "Right to privacy and informed consent"
            ]
          ]
        ]
        8 => array:2 [
          "identificador" => "sec0040"
          "titulo" => "Conflicts of interest"
        ]
        9 => array:1 [
          "titulo" => "References"
        ]
      ]
    ]
    "pdfFichero" => "main.pdf"
    "tienePdf" => true
    "fechaRecibido" => "2012-04-02"
    "fechaAceptado" => "2012-05-09"
    "PalabrasClave" => array:2 [
      "en" => array:1 [
        0 => array:4 [
          "clase" => "keyword"
          "titulo" => "Keywords"
          "identificador" => "xpalclavsec237851"
          "palabras" => array:3 [
            0 => "Scimitar syndrome"
            1 => "Abnormal pulmonary venous return"
            2 => "Coronary venous fistula"
          ]
        ]
      ]
      "pt" => array:1 [
        0 => array:4 [
          "clase" => "keyword"
          "titulo" => "Palavras-chave"
          "identificador" => "xpalclavsec237852"
          "palabras" => array:3 [
            0 => "S&#237;ndrome de Cimitarra"
            1 => "Drenagem pulmonar an&#243;mala"
            2 => "F&#237;stula coron&#225;ria"
          ]
        ]
      ]
    ]
    "tieneResumen" => true
    "resumen" => array:2 [
      "en" => array:2 [
        "titulo" => "Abstract"
        "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Scimitar syndrome &#40;SS&#41; is a rare congenital anomaly characterized by partial or complete anomalous pulmonary venous drainage of the right or left lung into the inferior vena cava&#46; The syndrome is commonly associated with hypoplasia of the right lung&#44; pulmonary sequestration&#44; persistent left superior vena cava&#44; and dextroposition of the heart&#46; We report a rare variant of SS in a 44-year-old man together with a single aortic trunk&#44; as well as a coronary-cameral venous fistula&#46;</p>"
      ]
      "pt" => array:2 [
        "titulo" => "Resumo"
        "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A s&#237;ndrome de Cimitarra &#40;SC&#41; &#233; uma malforma&#231;&#227;o cong&#233;nita rara&#44; caracterizada por uma drenagem pulmonar an&#243;mala parcial ou total do pulm&#227;o direito ou esquerdo&#44; na veia cava inferior&#46; Esta s&#237;ndrome &#233; frequentemente associada a hipoplasia do pulm&#227;o direito&#44; sequestra&#231;&#227;o pulmonar&#44; persist&#234;ncia de veia cava superior esquerda e dextroposi&#231;&#227;o do cora&#231;&#227;o&#46; Reportamos uma variante rara de SC num homem de 44 anos com tronco supra-a&#243;rtico &#250;nico e fistula coron&#225;rio-auricular&#46;</p>"
      ]
    ]
    "multimedia" => array:5 [
      0 => array:7 [
        "identificador" => "fig0005"
        "etiqueta" => "Figure 1"
        "tipo" => "MULTIMEDIAFIGURA"
        "mostrarFloat" => true
        "mostrarDisplay" => false
        "figura" => array:1 [
          0 => array:4 [
            "imagen" => "gr1.jpeg"
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        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Aortic arch angiography&#46; Takeoff of large vessels from the aortic arch via a single trunk &#40;white arrow&#41;&#46;</p>"
        ]
      ]
      1 => array:7 [
        "identificador" => "fig0010"
        "etiqueta" => "Figure 2"
        "tipo" => "MULTIMEDIAFIGURA"
        "mostrarFloat" => true
        "mostrarDisplay" => false
        "figura" => array:1 [
          0 => array:4 [
            "imagen" => "gr2.jpeg"
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        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Right upper pulmonary venous angiography&#46; Abnormal pulmonary venous drainage from the upper lobe of the right lung draining into the high SVC via the right upper pulmonary vein &#40;white arrow&#41;&#46;</p>"
        ]
      ]
      2 => array:7 [
        "identificador" => "fig0015"
        "etiqueta" => "Figure 3"
        "tipo" => "MULTIMEDIAFIGURA"
        "mostrarFloat" => true
        "mostrarDisplay" => false
        "figura" => array:1 [
          0 => array:4 [
            "imagen" => "gr3.jpeg"
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            "Ancho" => 945
            "Tamanyo" => 124547
          ]
        ]
        "descripcion" => array:1 [
          "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Right middle pulmonary venous angiography&#46; Abnormal pulmonary venous drainage from the middle lobe of the right lung draining into the high SVC via the right upper pulmonary vein &#40;white arrow&#41;&#46;</p>"
        ]
      ]
      3 => array:7 [
        "identificador" => "fig0020"
        "etiqueta" => "Figure 4"
        "tipo" => "MULTIMEDIAFIGURA"
        "mostrarFloat" => true
        "mostrarDisplay" => false
        "figura" => array:1 [
          0 => array:4 [
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Coronary venous-cameral fistula&#46; Using a 5-Fr MP diagnostic catheter&#44; angiography of the coronary-venous cameral fistula &#40;white arrow&#41; was performed&#46; The fistula itself drains into the high RA&#46;</p>"
        ]
      ]
      4 => array:7 [
        "identificador" => "fig0025"
        "etiqueta" => "Figure 5"
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        "mostrarDisplay" => false
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            "imagen" => "gr5.jpeg"
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        "descripcion" => array:1 [
          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Pulmonary angiography&#46; Using a 5-Fr pig tail catheter&#44; pulmonary angiography was performed&#44; which showed a moderately enlarged main PA&#46;</p>"
        ]
      ]
    ]
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      "titulo" => "References"
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        0 => array:2 [
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          "bibliografiaReferencia" => array:14 [
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                            0 => "P&#46;D&#46; Holt"
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                            0 => "Y&#46;A&#46; Gao"
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                    0 => array:2 [
                      "titulo" => "Scimitar vein anomaly with multiple cardiac malformations&#44; craniofacial&#44; and central nervous system abnormalities in a brother and sister&#58; familial scimitar anomaly or new syndrome&#63;"
                      "autores" => array:1 [
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
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                            0 => "C&#46;C&#46; Wang"
                            1 => "E&#46;T&#46; Wu"
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "A&#46; Rajaii-Khorasani"
                            1 => "M&#46; Kahrom"
                            2 => "H&#46; Mottaghi"
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                          ]
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:3 [
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                        0 => array:2 [
                          "etal" => false
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                            0 => "J&#46;R&#46; Morgan"
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                          "etal" => false
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                            1 => "M&#46;A&#46; Grant"
                            2 => "R&#46;A&#46; Gustafson"
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                      "autores" => array:1 [
                        0 => array:2 [
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                          ]
                        ]
                      ]
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                  ]
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                    0 => array:2 [
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                        "fecha" => "2011"
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                            "web" => "Medline"
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                  ]
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              "referencia" => array:1 [
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                  "contribucion" => array:1 [
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "B&#46; Uthaman"
                            1 => "L&#46; Abushaban"
                            2 => "M&#46; Al-Qbandi"
                            3 => "J&#46; Rathinasamy"
                          ]
                        ]
                      ]
                    ]
                  ]
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                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
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                            0 => "M&#46; Cilingiroglu"
                            1 => "A&#46; Hakeem"
                          ]
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Case report
Right-sided scimitar syndrome in a patient with a single aortic trunk and coronary-cameral venous fistula
Síndrome de Cimitarra direita num doente com truncus arteriosus e fístula venosa coronário-ventricular
Mehmet Cilingiroglua, Nuri Ilker Akkusb,
Autor para correspondência
iakkus@hotmail.com

Corresponding author.
a University of Pittsburgh Medical Center, Heart and Vascular Institute, Pittsburgh, United States
b LSU Shreveport Health Sciences Center, Shreveport, LA, United States
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but in one third&#44; this vein drains only the lower portion of the right lung&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Partial abnormalities of pulmonary venous return &#40;PAPVR&#41; are seen in 0&#46;4&#8211;0&#46;7&#37; of adult autopsies&#44; while patients with SS account for 3&#8211;5&#37; of these cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a> SS constitutes 0&#46;5&#8211;1&#37; of all congenital heart defects&#46; This rare anomaly has an incidence of approximately 1-3 per 100<span class="elsevierStyleHsp" style=""></span>000 live births&#59; however&#44; the true incidence may be higher because many patients remain asymptomatic&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The etiology is not completely understood&#46; In several patients with total anomalous pulmonary venous return&#44; the gene locus has been mapped to chromosome 4q12&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> and females are more frequently affected than males&#46; The scimitar sign refers to the crescent &#40;resembling the Turkish sword&#41; described by the descent of the anomalous pulmonary vein &#40;the tip of the crescent points inferiorly and medially to the diaphragm&#47;right heart border junction&#41;&#46; The concavity of the crescent is adjacent to the junction of the diaphragm and right heart border&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 44-year-old man presented with several weeks of palpitations&#46; He also described symptoms of dyspnea with mild exertion and substernal chest discomfort at rest&#46; He had a history of long-standing systemic hypertension&#46; The 12-lead ECG showed atrial fibrillation with rapid ventricular rate&#46; His 2D Doppler echocardiogram revealed a mildly enlarged RA and right ventricle &#40;RV&#41;&#46; He subsequently underwent right and left heart catheterization with selective coronary angiography&#46; Right and left heart catheterization with a full oximetry run to calculate shunts revealed Qp&#47;Qs of 1&#46;3&#46; Selective coronary angiography was performed using right radial artery access&#44; and showed no significant coronary disease&#46; However&#44; angiography of the aortic arch showed a single trunk takeoff for the large vessels from the aortic arch &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41;&#46; Using right femoral vein access&#44; a 5-Fr multipurpose diagnostic catheter was advanced into the upper &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41; and middle &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>&#41; right pulmonary veins as they opened into the superior vena cava &#40;SVC&#41;&#46; To exclude any possible associated atrial septal defect&#44; an MP-1 catheter was engaged into what proved to be a coronary-cameral fistula &#40;CCF&#41; opening into the RA separately from the coronary sinus &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Figure 4</a>&#41;&#44; the CCF went from the coronary vein to the RA and the coronary sinus was also filled with contrast retrogradely from the vein &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Figure 4</a>&#41;&#46; Pulmonary angiography using a 5-Fr pigtail catheter showed a moderately dilated pulmonary trunk &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Figure 5</a>&#41;&#46; Following consultations with the cardiothoracic surgery and pediatric cardiology teams&#44; it was felt that the best course of management would be to follow the patient clinically with serial echocardiography&#44; as there was no significant right-to-left shunt&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">SS is a complex form of PAPVR&#44; which is a connection failure between the right pulmonary veins and the LA during fetal development&#46; Variations in PAPVR include the right pulmonary veins draining into the SVC-LA junction&#44; RA&#44; or IVC&#44; or as in our case&#44; separately to a high SVC&#46; In SS&#44; an anomalous right pulmonary vein generally draining the entire right lung but occasionally the middle and superior lobes&#44; may descend in a cephalad-to-caudal direction toward the diaphragm with a crescent &#40;scimitar&#41; shape&#46; This vein then curves sharply to the left just above or below the IVC-RA junction&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> The anomalous right pulmonary venous trunk usually courses anterior to the hilum of the right lung and connects to the IVC just superior to the orifices of the hepatic veins&#46; Rarely&#44; it drains into the left atrium&#44; with normal venous return&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In this patient&#44; the right upper and middle pulmonary veins drained into the high SVC and then ultimately into the RA&#44; leading to increased pulmonary circulation as shown by the enlarged right chambers and main pulmonary artery&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The most common aortic arch branching pattern in humans consists of three great vessels originating from the arch of the aorta&#58; the innominate&#44; left common carotid and left subclavian arteries&#46; In &#8220;bovine&#8221; aortic arch&#44; rather than arising directly from the aortic arch as a separate branch&#44; the left common carotid artery origin is moved to the right and merges with the origin of the innominate artery&#44; or the left common carotid artery originates directly from the innominate artery rather than as a common trunk&#46; A single brachiocephalic trunk originating from the aortic arch and then dividing into right and left brachiocephalic trunks or trifurcating to brachiocephalic&#44; left common carotid and subclavian artery is a very rare anomaly&#44; with few cases reported in the literature&#44;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10&#44;11</span></a> and there is no previously reported case of SS with this anomaly&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Our patient had a venous CCF draining into the RA from the coronary vein in addition to the right upper and middle pulmonary veins draining through the SVC&#46; There is no previously reported case of SS associated with venous CCF in the literature&#46; Coronary-cameral fistulas are unusual congenital or acquired anomalous communications between an epicardial coronary artery and a cardiac chamber&#58; the RA&#44; coronary sinus&#44; right ventricle&#44; left atrium&#44; or left ventricle&#46; In a review of 304 patients with CCF and coronary vascular fistula &#40;CVF&#41;&#44; a continuous cardiac murmur was heard in 82&#37; of the subjects&#46; Dyspnea&#44; chest pain and angina pectoris&#44; palpitations&#44; and fatigue are reported symptoms in clinical presentations&#46; Although rare&#44; infective endocarditis has been reported in patients with CCF&#44; but not in those with CVF&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Surgical repair of SS consists of redirecting the pulmonary venous drainage into the LA&#44; by either baffling the anomalous drainage into the LA via a tunnel or transecting the &#8220;scimitar drainage&#8221; near its entrance into the IVC and then reimplanting it directly into the LA&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> The surgical option is&#44; however&#44; associated with a relatively high incidence of interruption failure&#44; reinserted pulmonary vein stenosis&#44; surgical complications&#44; and redo procedures&#46; Surgical correction is considered in symptomatic patients or in those with increased pulmonary blood flow and signs of right heart dilation with a significant shunt &#40;Qp&#47;Qs &#8805;1&#46;5&#41;&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In the most common form of SS&#44; there is anomalous right pulmonary vein drainage into the IVC with anomalous systemic arterial supply &#40;ASAS&#41; from the abdominal aorta toward the affected pulmonary parenchyma&#44; leading to pulmonary hypertension and subsequent volume overload and heart failure&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">If the presence of a large ASAS to the right lung with pulmonary overcirculation is detected preoperatively &#40;which it was not in our case&#41;&#44; occlusion of the ASAS is frequently performed percutaneously to encourage compensatory growth of the remaining normal lung tissue and to reduce both the associated left-to-right shunt and the risk of chronic and recurrent infection in the abnormal lung in later life&#46; Successful percutaneous occlusion of ASAS in infants with SS has emerged as an acceptable treatment using the Amplatzer&#8482; vascular plug device &#40;AGA Medical&#44; Golden Valley&#44; MN&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13&#44;14</span></a> In a study of 16 infants who underwent percutaneous ASAS interruption&#44; Uthaman et al&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> reported significant reduction in left-to-right shunt and pulmonary artery pressures in &#62;90&#37; of the patients&#44; which translated into symptomatic improvement as well as growth of the right lung in follow-up&#46; Furthermore&#44; this approach leads to sustained long-term clinical improvement without need for further surgical correction in the majority of patients&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; in this case report we describe a patient with scimitar syndrome with very unusual associated anomalies of a single aortic trunk and venous CCF&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Ethical disclosures</span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Protection of human and animal subjects</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Confidentiality of data</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data and that all the patients included in the study received sufficient information and gave their written informed consent to participate in the study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Right to privacy and informed consent</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article&#46; The corresponding author is in possession of this document&#46;</p></span></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflicts of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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          "titulo" => "Introduction"
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              "titulo" => "Protection of human and animal subjects"
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            1 => array:2 [
              "identificador" => "sec0030"
              "titulo" => "Confidentiality of data"
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            2 => array:2 [
              "identificador" => "sec0035"
              "titulo" => "Right to privacy and informed consent"
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    "tienePdf" => true
    "fechaRecibido" => "2012-04-02"
    "fechaAceptado" => "2012-05-09"
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          "clase" => "keyword"
          "titulo" => "Keywords"
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          "palabras" => array:3 [
            0 => "Scimitar syndrome"
            1 => "Abnormal pulmonary venous return"
            2 => "Coronary venous fistula"
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        ]
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            0 => "S&#237;ndrome de Cimitarra"
            1 => "Drenagem pulmonar an&#243;mala"
            2 => "F&#237;stula coron&#225;ria"
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        "titulo" => "Abstract"
        "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Scimitar syndrome &#40;SS&#41; is a rare congenital anomaly characterized by partial or complete anomalous pulmonary venous drainage of the right or left lung into the inferior vena cava&#46; The syndrome is commonly associated with hypoplasia of the right lung&#44; pulmonary sequestration&#44; persistent left superior vena cava&#44; and dextroposition of the heart&#46; We report a rare variant of SS in a 44-year-old man together with a single aortic trunk&#44; as well as a coronary-cameral venous fistula&#46;</p>"
      ]
      "pt" => array:2 [
        "titulo" => "Resumo"
        "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A s&#237;ndrome de Cimitarra &#40;SC&#41; &#233; uma malforma&#231;&#227;o cong&#233;nita rara&#44; caracterizada por uma drenagem pulmonar an&#243;mala parcial ou total do pulm&#227;o direito ou esquerdo&#44; na veia cava inferior&#46; Esta s&#237;ndrome &#233; frequentemente associada a hipoplasia do pulm&#227;o direito&#44; sequestra&#231;&#227;o pulmonar&#44; persist&#234;ncia de veia cava superior esquerda e dextroposi&#231;&#227;o do cora&#231;&#227;o&#46; Reportamos uma variante rara de SC num homem de 44 anos com tronco supra-a&#243;rtico &#250;nico e fistula coron&#225;rio-auricular&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Aortic arch angiography&#46; Takeoff of large vessels from the aortic arch via a single trunk &#40;white arrow&#41;&#46;</p>"
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Right upper pulmonary venous angiography&#46; Abnormal pulmonary venous drainage from the upper lobe of the right lung draining into the high SVC via the right upper pulmonary vein &#40;white arrow&#41;&#46;</p>"
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          "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Right middle pulmonary venous angiography&#46; Abnormal pulmonary venous drainage from the middle lobe of the right lung draining into the high SVC via the right upper pulmonary vein &#40;white arrow&#41;&#46;</p>"
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Coronary venous-cameral fistula&#46; Using a 5-Fr MP diagnostic catheter&#44; angiography of the coronary-venous cameral fistula &#40;white arrow&#41; was performed&#46; The fistula itself drains into the high RA&#46;</p>"
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Pulmonary angiography&#46; Using a 5-Fr pig tail catheter&#44; pulmonary angiography was performed&#44; which showed a moderately enlarged main PA&#46;</p>"
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Informação do artigo
ISSN: 08702551
Idioma original: Inglês
Dados atualizados diariamente
Ano/Mês Html Pdf Total
2024 Novembro 11 8 19
2024 Outubro 62 50 112
2024 Setembro 82 34 116
2024 Agosto 80 53 133
2024 Julho 53 36 89
2024 Junho 44 33 77
2024 Maio 54 34 88
2024 Abril 56 34 90
2024 Maro 55 25 80
2024 Fevereiro 47 29 76
2024 Janeiro 57 31 88
2023 Dezembro 43 34 77
2023 Novembro 57 33 90
2023 Outubro 34 24 58
2023 Setembro 45 33 78
2023 Agosto 34 18 52
2023 Julho 29 15 44
2023 Junho 47 14 61
2023 Maio 68 31 99
2023 Abril 46 9 55
2023 Maro 37 24 61
2023 Fevereiro 41 25 66
2023 Janeiro 36 19 55
2022 Dezembro 34 31 65
2022 Novembro 48 27 75
2022 Outubro 48 30 78
2022 Setembro 29 29 58
2022 Agosto 41 36 77
2022 Julho 51 35 86
2022 Junho 29 25 54
2022 Maio 31 27 58
2022 Abril 36 26 62
2022 Maro 23 32 55
2022 Fevereiro 26 18 44
2022 Janeiro 32 17 49
2021 Dezembro 29 34 63
2021 Novembro 48 35 83
2021 Outubro 41 49 90
2021 Setembro 39 38 77
2021 Agosto 42 29 71
2021 Julho 50 21 71
2021 Junho 55 21 76
2021 Maio 39 33 72
2021 Abril 48 40 88
2021 Maro 68 18 86
2021 Fevereiro 58 10 68
2021 Janeiro 43 10 53
2020 Dezembro 44 11 55
2020 Novembro 49 13 62
2020 Outubro 40 15 55
2020 Setembro 60 12 72
2020 Agosto 21 12 33
2020 Julho 57 6 63
2020 Junho 40 12 52
2020 Maio 59 5 64
2020 Abril 42 23 65
2020 Maro 56 12 68
2020 Fevereiro 115 24 139
2020 Janeiro 46 14 60
2019 Dezembro 42 17 59
2019 Novembro 39 7 46
2019 Outubro 52 9 61
2019 Setembro 26 12 38
2019 Agosto 33 11 44
2019 Julho 51 14 65
2019 Junho 52 13 65
2019 Maio 49 32 81
2019 Abril 40 23 63
2019 Maro 106 30 136
2019 Fevereiro 101 17 118
2019 Janeiro 48 6 54
2018 Dezembro 72 11 83
2018 Novembro 97 9 106
2018 Outubro 203 31 234
2018 Setembro 78 14 92
2018 Agosto 44 14 58
2018 Julho 21 8 29
2018 Junho 47 6 53
2018 Maio 80 10 90
2018 Abril 52 6 58
2018 Maro 91 6 97
2018 Fevereiro 27 5 32
2018 Janeiro 31 3 34
2017 Dezembro 59 13 72
2017 Novembro 42 12 54
2017 Outubro 40 19 59
2017 Setembro 37 7 44
2017 Agosto 54 8 62
2017 Julho 36 10 46
2017 Junho 65 13 78
2017 Maio 40 7 47
2017 Abril 31 3 34
2017 Maro 32 26 58
2017 Fevereiro 33 8 41
2017 Janeiro 30 9 39
2016 Dezembro 34 6 40
2016 Novembro 34 6 40
2016 Outubro 67 7 74
2016 Setembro 28 7 35
2016 Agosto 28 2 30
2016 Julho 13 3 16
2016 Junho 4 0 4
2016 Maio 14 5 19
2016 Abril 16 4 20
2016 Maro 45 8 53
2016 Fevereiro 68 13 81
2016 Janeiro 54 14 68
2015 Dezembro 53 7 60
2015 Novembro 55 3 58
2015 Outubro 68 14 82
2015 Setembro 68 12 80
2015 Agosto 67 18 85
2015 Julho 37 3 40
2015 Junho 34 0 34
2015 Maio 59 8 67
2015 Abril 63 10 73
2015 Maro 45 5 50
2015 Fevereiro 53 8 61
2015 Janeiro 44 8 52
2014 Dezembro 57 11 68
2014 Novembro 52 11 63
2014 Outubro 88 19 107
2014 Setembro 53 10 63
2014 Agosto 54 17 71
2014 Julho 57 12 69
2014 Junho 63 6 69
2014 Maio 56 6 62
2014 Abril 52 9 61
2014 Maro 73 17 90
2014 Fevereiro 74 13 87
2014 Janeiro 78 13 91
2013 Dezembro 65 17 82
2013 Novembro 56 16 72
2013 Outubro 65 8 73
2013 Setembro 71 21 92
2013 Agosto 74 23 97
2013 Julho 112 26 138
2013 Junho 65 14 79
2013 Maio 71 15 86
2013 Abril 90 32 122
2013 Maro 70 38 108
2013 Fevereiro 53 40 93
2013 Janeiro 42 19 61
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