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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">The Fontan procedure is the final step in the staged surgical palliation of patients with a functionally univentricular heart&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> It results in the creation of a circulation in series in which the systemic venous blood is diverted to the pulmonary arteries&#44; bypassing the subpulmonary ventricle&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">1&#44;2</span></a> In order for the Fontan circuit to be achievable&#44; there must be a low resistance to blood flow throughout the circuit&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Various modifications of the original Fontan procedure have been devised&#46; One of them is the lateral tunnel Fontan&#44; which involves construction of an intra-atrial baffle to direct the inferior vena caval blood to the lungs&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> In addition&#44; a small fenestration can be made in the baffle to provide preload to the systemic ventricle and decompress the systemic venous pathway&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Protein-losing enteropathy refers to excessive protein loss into the gastrointestinal tract&#44; leading to the development of edema&#44; ascites&#44; pleural effusions&#44; and other complications&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">2</span></a> Clinical presentation and laboratory findings&#44; including low serum albumin levels and increased fecal clearance of alpha-1-antitrypsin&#44; help establish the diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">2</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 26-year-old female patient presented with a three-week history of increasing dyspnea on exertion&#44; swelling of the legs&#44; and periorbital puffiness that was worse on awakening&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was known to have had double outlet right ventricle&#44; ventricular septal defect&#44; and severe mitral stenosis since birth&#46; She had undergone an atrial septectomy and pulmonary artery banding at one year of age&#46; At the age of 6&#46;5 years&#44; she had had a lateral tunnel Fontan procedure using a size 14 Gore-Tex tube graft with a fenestration&#44; constructed employing Laks&#8217; technique for creating a so-called adjustable atrial septal defect&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">3</span></a> The postoperative course had been uneventful&#46; She had been taking prophylactic acenocoumarol&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">On physical examination&#44; there was mild central cyanosis with oxygen saturation of 85&#37; on room air and clubbing&#46; She had mild periorbital and marked pretibial&#44; ankle&#44; and foot edema&#46; The abdomen was slightly distended&#44; and the liver was palpable 3 cm below the right costal margin&#46; Peripheral pulses were normal&#44; and blood pressure was 110&#47;70 mmHg&#46; The heart rate was 98 beats per minute and regular&#46; There was a single second sound and a faint systolic murmur over the left upper sternal border&#46; The lungs were clear on auscultation&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The electrocardiogram showed sinus rhythm&#44; normal axis&#44; right ventricular dominance&#44; and incomplete right bundle branch block&#46; The chest X-ray demonstrated mild cardiomegaly with normal pulmonary vascular markings and clear lung fields&#46; Blood chemistry was notable for decreased total protein and albumin levels &#40;4&#46;9 and 2&#46;4 g&#47;dl&#44; respectively&#41;&#46; Twenty-four-hour fecal alpha-1-antitrypsin clearance was significantly elevated &#40;300&#46;8 ml&#47;24 hours&#41;&#44; leading to a diagnosis of protein-losing enteropathy&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Transthoracic echocardiography confirmed the above cardiac diagnosis&#46; It also showed a restrictive atrial septal defect &#40;ASD&#41; measuring 4 mm in diameter&#44; with a peak flow velocity of 2&#46;3 m&#47;s across it&#46; The left atrium was slightly dilated&#44; while the left ventricle was moderately hypoplastic&#46; The mitral valve leaflets appeared dysplastic and thickened&#44; with diastolic doming&#46; The mitral valve orifice area was 8&#46;5 mm<span class="elsevierStyleSup">2</span>&#46; These findings were further confirmed by cardiac magnetic resonance imaging &#40;MRI&#41; &#40;Supplementary Video 1&#41;&#46; Both left ventricular end-diastolic volume and left ventricular mass were significantly decreased&#46; The right ventricular cardiac index was calculated to be 2&#46;1 l&#47;min&#47;m<span class="elsevierStyleSup">2</span>&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Cardiac catheterization was undertaken to widen the interatrial communication&#46; The patient received intravenous heparin at a dose of 50 IU&#47;kg and prophylactic ceftriaxone before the procedure&#46; After obtaining right femoral vein access&#44; a catheter was introduced into the inferior vena cava &#40;IVC&#41;&#46; The central venous pressure was 16&#47;14 with a mean of 15 mmHg&#46; There was a mean pressure gradient of 1 mmHg from the IVC to the left pulmonary artery&#46; No pressure gradient was measured from the IVC to the right pulmonary artery&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">There was a small right-to-left shunt through the 2&#46;3 mm wide fenestration between the lateral tunnel and the left atrium &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41;&#46; The catheter was passed into the left atrium through the fenestration&#46; The mean left atrial pressure was measured at 15 mmHg&#46; Intraprocedural transesophageal echocardiography &#40;TEE&#41; revealed a restrictive ASD about 3 mm in diameter with a peak flow velocity of 2&#46;2 m&#47;s &#40;pressure gradient of about 20 mmHg&#41; across it&#46; A guide wire was then advanced from the left atrium across the ASD into the right atrium&#46; The mean right atrial pressure was 11 mmHg&#46; Next&#44; a snare catheter was passed into the aorta via the right femoral artery&#44; and advanced into the right ventricle and the right atrium&#46; The guide wire in the right atrium was grasped by the snare catheter&#44; thus creating an arteriovenous loop&#46; A 20 mm &#215; 45 mm balloon catheter &#40;Balton&#44; Warsaw&#44; Poland&#41; was subsequently inserted over this arteriovenous loop and the balloon was inflated within the ASD&#46; However&#44; the balloon ruptured while fully inflated&#46; The widened ASD now measured about 6 mm in diameter&#46; There was a peak flow velocity of 1&#46;6 m&#47;s &#40;pressure gradient of about 10 mmHg&#41; across the defect as demonstrated by TEE&#46; This result was deemed unsatisfactory and it was decided to repeat balloon dilation with a larger balloon&#46; The catheter was again introduced and the guide wire was passed through the lateral tunnel&#44; fenestration&#44; left atrium&#44; and the ASD into the right atrium&#46; A 25 mm &#215; 40 mm balloon catheter &#40;Osypka AG&#44; Rheinfelden-Herten&#44; Germany&#41; was advanced over the guide wire and positioned across the ASD &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>A&#41;&#46; The balloon was inflated until disappearance of the waist on the balloon &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>B&#41;&#46; After the septum was torn&#44; the mean pressure gradient between the atria fell from 4 to 2 mmHg&#46; TEE showed a final defect size of 11 mm with laminar flow across it &#40;up to 1&#46;2 m&#47;s&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">Follow-up cardiac MRI demonstrated an unrestrictive atrial septum &#40;Supplementary Video 2&#41;&#46; In addition&#44; the right ventricular cardiac index increased to 3&#46;2 l&#47;min&#47;m<span class="elsevierStyleSup">2</span>&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The signs and symptoms of protein-losing enteropathy gradually subsided after the intervention&#46; The patient has been doing well for three years now&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0065" class="elsevierStylePara elsevierViewall">Protein-losing enteropathy is a relatively rare complication of the Fontan circulation&#59; however&#44; it is associated with significant morbidity and mortality&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">2&#44;4</span></a> Proposed underlying etiologies include impaired hemodynamics &#40;particularly diminished cardiac output&#41;&#44; increased mesenteric vascular resistance&#44; systemic inflammation&#44; and altered glycosaminoglycan content in the basal membrane of enterocytes&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> Protein-losing enteropathy without an established cause remains difficult to treat&#44; despite improvements in medical therapy&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">4</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">The diagnosis of protein-losing enteropathy in a Fontan patient necessitates careful investigation for the presence of an underlying hemodynamic disturbance that can be managed&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">2</span></a> Improvements in protein-losing enteropathy have been reported after surgical revision of the circuit&#44; interventional procedures &#40;including percutaneous Fontan fenestrations and balloon dilations with or without stent placement&#41;&#44; cardiac pacing&#44; and cardiac transplantation&#46;<a class="elsevierStyleCrossRefs" href="#bib0105"><span class="elsevierStyleSup">6&#8211;10</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">In the presence of an atretic or stenotic atrioventricular valve&#44; pulmonary venous outflow has to cross the atrial septum to reach the systemic ventricle&#46;<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">11&#44;12</span></a> In such cases&#44; it is crucial to create a non-restrictive interatrial communication at the first stage of palliation&#46; Rarely&#44; if the surgical resection of the atrial septum is inadequate or the rims of the defect become thickened and scarred&#44; obstruction of pulmonary venous outflow at the atrial septal level may occur&#46;<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">13&#44;14</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Pulmonary venous outflow obstruction is commonly manifested by persistent pleural and pericardial effusions&#44; ascites&#44; and heart failure&#46;<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">15</span></a> Full-blown protein-losing enteropathy may also develop&#46;<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">11&#44;15&#44;16</span></a> The diagnosis is usually made by echocardiography&#46; Cardiac MRI may also be helpful&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">2&#44;12&#44;15</span></a> Cardiac catheterization is performed to obtain hemodynamic data and potentially address hemodynamic abnormalities&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">2&#44;16</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">Obstruction to flow across the residual atrial septum has traditionally been treated by surgical excision of the atrial septum and unroofing of the coronary sinus&#46;<a class="elsevierStyleCrossRefs" href="#bib0135"><span class="elsevierStyleSup">12&#44;13</span></a> To our knowledge&#44; however&#44; there is only one description of percutaneous relief of the obstruction across the atrial septum after the Fontan procedure&#44;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">16</span></a> although this might be the preferred treatment modality&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0090" class="elsevierStylePara elsevierViewall">Protein-losing enteropathy in patients with Fontan circulation and atretic or stenotic atrioventricular valve may rarely result from obstruction to pulmonary venous outflow at the atrial septal level&#46; In this setting&#44; percutaneous enlargement of the restrictive ASD may be a safe and feasible alternative to surgical resection&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflicts of interest</span><p id="par0095" 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" => "Case report"
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          "titulo" => "Discussion"
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          "titulo" => "Conclusion"
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          "identificador" => "sec0025"
          "titulo" => "Conflicts of interest"
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          "titulo" => "References"
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    "fechaRecibido" => "2018-03-02"
    "fechaAceptado" => "2018-09-30"
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          "clase" => "keyword"
          "titulo" => "Keywords"
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          "palabras" => array:6 [
            0 => "Fontan procedure"
            1 => "Protein-losing enteropathy"
            2 => "Atrial septum"
            3 => "Cardiac catheterization"
            4 => "Mitral stenosis"
            5 => "Double outlet right ventricle"
          ]
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        0 => array:4 [
          "clase" => "keyword"
          "titulo" => "Palavras-chave"
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          "palabras" => array:6 [
            0 => "Procedimento de Fontan"
            1 => "Enteropatia por perda de prote&#237;nas"
            2 => "Septo auricular"
            3 => "Cateterismo card&#237;aco"
            4 => "Estenose mitral"
            5 => "Dupla sa&#237;da do ventr&#237;culo direito"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Protein-losing enteropathy is one of the most feared complications of the Fontan circulation&#46; The diagnosis of protein-losing enteropathy in this setting should prompt a thorough investigation for the presence of a treatable hemodynamic impairment&#46; In this report&#44; we describe a complete reversal of protein-losing enteropathy following percutaneous enlargement of a restrictive atrial septal defect in a patient with a fenestrated lateral tunnel Fontan and severe mitral stenosis&#46;</p></span>"
      ]
      "pt" => array:2 [
        "titulo" => "Resumo"
        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A enteropatia por perda de prote&#237;nas &#233; uma das complica&#231;&#245;es mais temidas da circula&#231;&#227;o de Fontan&#46; O diagn&#243;stico de enteropatia por perda de prote&#237;nas neste contexto deve suscitar uma avalia&#231;&#227;o exaustiva da eventual presen&#231;a de uma altera&#231;ao hemodin&#226;mica trat&#225;vel&#46; Neste artigo&#44; apresentamos uma completa regress&#227;o de enteropatia por perda de prote&#237;nas ap&#243;s dilata&#231;&#227;o percut&#226;nea de defeito restritivo do septo interauricular num doente com tunel lateral fenestrado de Fontan e estenose mitral grave&#46;</p></span>"
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            "apendice" => "<p id="par0105" class="elsevierStylePara elsevierViewall">The following are the supplementary material to this article&#58;<elsevierMultimedia ident="upi0005"></elsevierMultimedia><elsevierMultimedia ident="upi0010"></elsevierMultimedia></p>"
            "etiqueta" => "Appendix A"
            "titulo" => "Supplementary data"
            "identificador" => "sec0035"
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          "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Venography from the inferior vena cava shows the lateral tunnel &#40;LT&#41; total cavopulmonary connection&#46; There is also a fenestration in the lateral tunnel opening into the left atrium &#40;arrow&#41;&#46;</p>"
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Balloon dilation of a restrictive atrial septal defect &#40;ASD&#41;&#46; &#40;A&#41; A waist on the balloon at the site of the restrictive ASD &#40;arrowheads&#41;&#59; &#40;B&#41; the waist was completely abolished after full inflation of the balloon&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Supplementary Video 1</span> Cardiac magnetic resonance imaging showing a restrictive atrial septal defect&#44; the lateral tunnel with a fenestration opening into a mildly dilated left atrium&#44; and stenotic mitral valve&#46;</p>"
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Supplementary Video 2</span> Follow-up cardiac magnetic resonance imaging demonstrating unrestrictive atrial septum&#46;</p>"
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