Case report
Anomalous origin of left coronary artery: A malignant interarterial variant with a benign clinical course
Origem anómala da coronária esquerda: variante interarterial maligna com evolução clínica benigna
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"etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "pt" => array:1 [ "titulo" => "Origem anómala da coronária esquerda: variante interarterial maligna com evolução clínica benigna" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 951 "Ancho" => 1583 "Tamanyo" => 113758 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Multiplanar reconstruction. Ao: aorta; PA: pulmonary artery; LM: left main.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Coronary artery anomalies are the third leading cause of sudden death in young athletes<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a>; their incidence in healthy individuals is estimated at 0.3–1%.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Various different anomalies have been described, and classifications have been proposed based on anatomy (anomalies of origin, course and termination) and on prognosis (with or without clinical significance).<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> With regard to the latter, most anomalies have no clinical significance and thus require no specific treatment.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">However, congenital coronary artery fistulas, myocardial bridging and anomalous origin in the pulmonary artery can be associated with malignant evolution.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Another potentially serious anomaly is origin of a coronary artery from the contralateral coronary sinus, which can lead to myocardial ischemia and sudden death when it courses between the aorta and pulmonary artery (interarterial course). Other courses—retroaortic, prepulmonary and subpulmonary, and septal—are considered benign.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Origin of the left coronary artery in the right sinus of Valsalva is rare, with an estimated incidence of 0.09–0.11% in patients undergoing coronary angiography,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> but it is frequently associated with sudden cardiac death during intense physical activity and in adolescents and young adults, half of whom were previously asymptomatic.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a> An interarterial course is seen in most of these patients<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a>; their poor prognosis may be due to the acute angle of the artery from the ostium, stretching of the intramural segment, or compression between the commissure of the right and left coronary cusps.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Cardiac CT angiography is a recent and valuable complementary exam that has high spatial resolution and provides three-dimensional views of the coronary arteries, with better definition of their origin and proximal portion, course, and relations with other cardiac structures than conventional angiography.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> The increasing popularity of this technique has greatly improved detection of coronary artery anomalies, which has made clinical decision-making more difficult, since there is, and is unlikely to be, sufficient evidence on which to base therapeutic strategies in doubtful cases such as the one presented.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case report</span><p id="par0035" class="elsevierStylePara elsevierViewall">A 54-year-old male patient was assessed in the cardiology outpatient clinic for atypical chest pain of about one year's evolution. He reported no other relevant cardiovascular symptoms, including fatigue, dyspnea, palpitations or syncope.</p><p id="par0040" class="elsevierStylePara elsevierViewall">His cardiovascular risk factors included hypertension and dyslipidemia; he also had congenital left leg atrophy. He was medicated with simvastatin, propanolol, candesartan and aspirin.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The electrocardiogram showed sinus rhythm with incomplete right bundle branch block but no other significant alterations. He had undergone myocardial perfusion scintigraphy with adenosine a year before, which was negative for ischemia.</p><p id="par0050" class="elsevierStylePara elsevierViewall">To clarify the clinical picture and since he was considered to have a low to intermediate likelihood of coronary artery disease, and also bearing in mind his physical limitations, he was referred for cardiac CT to rule out coronary artery disease. The exam showed a left coronary artery arising from the right coronary cusp and with an interarterial course, between the aorta and pulmonary trunk (<a class="elsevierStyleCrossRefs" href="#fig0005">Figures 1–3</a>). No coronary plaque was detected and his calcium score was 0.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">Given the clinical context of his age (54 years) and absence of typical angina or syncope, it was decided to perform ischemia testing. Although imaging exams such as dobutamine stress echocardiography, myocardial perfusion scintigraphy and magnetic resonance imaging have higher sensitivity and specificity for ischemia testing, treadmill exercise testing was chosen in order to determine the patient's actual maximum functional capacity. Beta-blocker therapy was suspended for the test.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The patient achieved maximal exercise (3<span class="elsevierStyleHsp" style=""></span>min 12<span class="elsevierStyleHsp" style=""></span>s with the Bruce protocol), reaching the maximum predicted heart rate with no angina or ST-segment alterations suggestive of ischemia, and with no arrhythmias. In the light of this result, it was decided to keep the patient under clinical surveillance with no referral for cardiac surgery.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0065" class="elsevierStylePara elsevierViewall">Although there is still disagreement on the subject, some guidelines recommend surgical coronary revascularization in patients with anomalous left coronary artery arising from the right sinus of Valsalva and coursing between the aorta and pulmonary artery (class I recommendation, level of evidence B in the ACC/AHA guidelines for the management of adults with congenital heart disease).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> When the right coronary has an anomalous origin, ischemia testing is recommended before referral for surgical correction (class I recommendation, level of evidence B).</p><p id="par0070" class="elsevierStylePara elsevierViewall">However, this case had some unusual aspects: the patient was older than the age-group most affected; he had no symptoms that were unequivocally associated with anomalous origin of the coronary arteries, particularly typical exertional angina or history of syncope; and no ischemia was documented on exercise testing, in which the patient achieved the limit of his functional capacity (maximum predicted heart rate) without the effect of beta-blockers. The patient's congenital left leg atrophy may paradoxically have had a protective effect by preventing him from performing strenuous exercise, which can trigger malignant arrhythmias in this situation.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Therefore, considering all factors, it was decided that the risk-benefit ratio favored a conservative approach.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflicts of interest</span><p id="par0080" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "xres176222" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec164592" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres176223" "titulo" => "Resumo" ] 3 => array:2 [ "identificador" => "xpalclavsec164591" "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:2 [ "identificador" => "sec0020" "titulo" => "Conflicts of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2011-10-03" "fechaAceptado" => "2011-11-30" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec164592" "palabras" => array:3 [ 0 => "Anomalous origin of coronary arteries" 1 => "Cardiac CT" 2 => "Sudden death" ] ] ] "pt" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palavras-chave" "identificador" => "xpalclavsec164591" "palabras" => array:3 [ 0 => "Origem anómala das coronárias" 1 => "AngioTC cardíaca" 2 => "Morte súbita" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Anomalous origin of coronary arteries represents a clinical challenge because of the anatomical variability and possible functional consequences, the pathophysiological mechanisms involved, and the lack of large published series that would provide evidence to guide the clinical and therapeutic approach. The authors describe the case of a 55-year-old male patient with a long history of atypical chest pain who was considered to have a low to intermediate likelihood of coronary artery disease. Therefore, and also bearing in mind his physical limitations (congenital left leg atrophy), he was referred for cardiac CT to rule out coronary artery disease. The exam showed a left coronary artery arising from the right coronary cusp and with an interarterial course, between the aorta and pulmonary trunk. Although this is a potentially malignant anatomical variant with surgical indication, a conservative approach was chosen, considering the late diagnosis and particular risk-benefit profile.</p>" ] "pt" => array:2 [ "titulo" => "Resumo" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A origem anómala das artérias coronárias constitui um importante desafio clínico pela variabilidade anatómica, possíveis repercussões funcionais, mecanismos fisiopatológicos implicados e também pela ausência de grandes séries na literatura que forneçam sólida evidência científica para a sua orientação clínica e terapêutica.</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Os autores descrevem o caso de um doente de 55 anos, com antecedentes de hipertensão, dislipidemia e atrofia congénita da perna esquerda, que se apresentou com quadro de dor retroesternal atípica com um ano de evolução. Tendo em conta os fatores de risco cardiovasculares e exames complementares de diagnóstico previamente realizados, considerou-se ser um doente com probabilidade baixa a intermédia de doença coronária. Assim, e pela sua limitação funcional, realizou angioTC cardíaca que revelou uma origem anómala da coronária esquerda na cúspide coronária direita e com trajeto entre a aorta e a artéria pulmonar. Embora esta seja uma variante anatómica com potencial evolução maligna, optou-se por uma atitude conservadora após ponderação do risco-benefício no contexto clínico deste doente.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Carvalho, S., et al. Origem anómala da coronária esquerda: variante interarterial maligna com evolução clínica benigna. Rev Port Cardiol 2012. <span class="elsevierStyleInterRef" href="doi:10.1016/j.repc.2011.11.010">doi:10.1016/j.repc.2011.11.010</span></p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 951 "Ancho" => 1583 "Tamanyo" => 113758 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Multiplanar reconstruction. Ao: aorta; PA: pulmonary artery; LM: left main.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1132 "Ancho" => 1583 "Tamanyo" => 129761 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Multiplanar reconstruction. Ao: aorta; PA: pulmonary artery; LCA: left coronary artery.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1062 "Ancho" => 2000 "Tamanyo" => 183507 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Volume-rendering technique. Ao: aorta; RVOT: right ventricular outflow tract; LM: left main.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Sudden death in young athletes" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "B.J. 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| 2018 September | 46 | 12 | 58 |
| 2018 August | 43 | 6 | 49 |
| 2018 July | 124 | 0 | 124 |
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| 2017 July | 45 | 15 | 60 |
| 2017 June | 79 | 10 | 89 |
| 2017 May | 159 | 11 | 170 |
| 2017 April | 64 | 9 | 73 |
| 2017 March | 70 | 24 | 94 |
| 2017 February | 48 | 6 | 54 |
| 2017 January | 51 | 7 | 58 |
| 2016 December | 109 | 21 | 130 |
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| 2016 October | 161 | 17 | 178 |
| 2016 September | 132 | 19 | 151 |
| 2016 August | 26 | 2 | 28 |
| 2016 July | 59 | 9 | 68 |
| 2016 June | 35 | 10 | 45 |
| 2016 May | 35 | 7 | 42 |
| 2016 April | 132 | 2 | 134 |
| 2016 March | 159 | 25 | 184 |
| 2016 February | 172 | 29 | 201 |
| 2016 January | 157 | 24 | 181 |
| 2015 December | 173 | 21 | 194 |
| 2015 November | 184 | 16 | 200 |
| 2015 October | 195 | 18 | 213 |
| 2015 September | 182 | 19 | 201 |
| 2015 August | 312 | 15 | 327 |
| 2015 July | 196 | 12 | 208 |
| 2015 June | 138 | 6 | 144 |
| 2015 May | 133 | 8 | 141 |
| 2015 April | 160 | 14 | 174 |
| 2015 March | 143 | 5 | 148 |
| 2015 February | 121 | 11 | 132 |
| 2015 January | 106 | 8 | 114 |
| 2014 December | 130 | 22 | 152 |
| 2014 November | 121 | 12 | 133 |
| 2014 October | 179 | 15 | 194 |
| 2014 September | 181 | 17 | 198 |
| 2014 August | 129 | 8 | 137 |
| 2014 July | 130 | 20 | 150 |
| 2014 June | 140 | 5 | 145 |
| 2014 May | 135 | 16 | 151 |
| 2014 April | 143 | 7 | 150 |
| 2014 March | 146 | 13 | 159 |
| 2014 February | 151 | 15 | 166 |
| 2014 January | 185 | 17 | 202 |
| 2013 December | 170 | 9 | 179 |
| 2013 November | 167 | 20 | 187 |
| 2013 October | 158 | 17 | 175 |
| 2013 September | 145 | 15 | 160 |
| 2013 August | 140 | 18 | 158 |
| 2013 July | 122 | 27 | 149 |
| 2013 June | 127 | 15 | 142 |
| 2013 May | 125 | 19 | 144 |
| 2013 April | 126 | 36 | 162 |
| 2013 March | 92 | 17 | 109 |
| 2013 February | 87 | 25 | 112 |
| 2013 January | 104 | 28 | 132 |
| 2012 December | 94 | 16 | 110 |
| 2012 November | 78 | 17 | 95 |
| 2012 October | 51 | 19 | 70 |
| 2012 September | 19 | 5 | 24 |
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