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extramural&#44; and straight&#46; Furthermore&#44; the structure of an intercoronary arterial connection is typical of an epicardial coronary artery&#44; with a well-defined muscular layer&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Coronary artery fistulas are abnormal communications between a coronary artery and a cardiac chamber or major vessel&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> They may be congenital or acquired due to trauma or iatrogenic causes&#46; Angiographic series reveal an incidence of coronary artery fistula in adults of 0&#46;3&#8211;0&#46;8&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#8211;6</span></a> Most of these patients are asymptomatic&#44; but heart failure&#44; angina&#44; myocardial infarction&#44; coronary steal&#44; endocarditis&#44; and dyspnea have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We report an interesting case of congenital coronary artery fistula in an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery presenting as an acute coronary syndrome&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">To the best of our knowledge&#44; this is the first case in the literature involving a coronary artery fistula in an intercoronary communication&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0025" class="elsevierStylePara elsevierViewall">A 62-year-old man was admitted to our hospital with sudden development of general weakness&#44; dizziness and a sensation of compression in his chest&#46; He had a history of diabetes mellitus&#46; At presentation his blood pressure was 80&#47;40 mmHg and heart rate was 65 beats&#47;min&#46; The ECG revealed sinus rhythm and 1&#8211;2 mm ST elevation in the anterior leads &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41;&#46; The patient was taken to the catheterization laboratory for percutaneous coronary intervention&#46; The left main and left circumflex coronary arteries were normal&#46; Coronary angiography showed a communication between the left main and the diagonal branch of the left anterior descending and a fistula between the intercoronary connection and the left atrium &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41;&#46; The other coronary arteries were normal&#46; Laboratory test results&#44; including cardiac troponin I and creatine kinase&#8211;MB levels&#44; were normal&#46; The angina symptoms disappeared and the ST elevation resolved within four hours &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>&#41;&#46; To the best of our knowledge&#44; this is the first case in the literature involving a congenital coronary artery fistula in an intercoronary communication&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">The hemodynamic consequences of coronary artery fistulas are variable depending on shunt size&#44; site of the shunt and presence of other underlying heart disease&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Fifty percent of patients with large or multiple fistulas may develop complications&#44; which may include bacterial endocarditis&#44; thrombosis&#44; aneurysm formation&#44; dissection&#44; rupture&#44; premature atherosclerosis&#44; pulmonary hypertension&#44; myocardial ischemia&#44; or infarction&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The functional significance of large intercoronary communications between normal coronary arteries is unclear but one may speculate that they have a potential role in protecting the myocardium should significant atherosclerosis develop in either of the patent arteries<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a>&#59; on the other hand&#44; myocardial ischemia could result if an unidirectional intercoronary communication causes a coronary steal phenomenon that results in inadequate perfusion&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> The ischemic consequences of an intercoronary connection with unidirectional flow may be explained by its similarity to a fistula from a coronary artery to a low-pressure cardiac space&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">We believe that an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery and a congenital coronary artery fistula may be the cause of ischemia in this case&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Ethical disclosures</span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Protection of human and animal subjects</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that the procedures followed were in accordance with the regulations of the relevant clinical research ethics committee and with those of the Code of Ethics of the World Medical Association &#40;Declaration of Helsinki&#41;&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Confidentiality of data</span><p id="par0050" 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" id="sect0055">Right to privacy and informed consent</span><p id="par0055" 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" id="sect0060">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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              "titulo" => "Protection of human and animal subjects"
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        "titulo" => "Abstract"
        "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Intercoronary communication is a very rare coronary artery anomaly&#46; It is defined as an open-ended circulation with bidirectional blood flow between two coronary arteries&#46; Coronary artery fistulas are abnormal communications between a coronary artery and a cardiac chamber or major vessel&#46; A 62-year-old man was admitted to our hospital with sudden development of general weakness&#44; dizziness and a sensation of compression in his chest&#46; At presentation his blood pressure was 80&#47;40 mmHg and heart rate was 65 beats&#47;min&#46; The ECG revealed sinus rhythm and 1&#8211;2 mm ST elevation in the anterior leads&#46; The patient was taken to the catheterization laboratory for percutaneous coronary intervention&#46; The left main and left circumflex coronary arteries were normal&#46; Coronary angiography showed a communication between the left main and the diagonal branch of the left anterior descending and a fistula between the intercoronary connection and the left atrium&#46; The other coronary arteries were normal&#46; Laboratory test results&#44; including cardiac troponin I and creatine kinase&#8211;MB levels&#44; were normal&#46; The angina symptoms disappeared and the ST elevation resolved within four hours&#46; We report an interesting case of congenital coronary artery fistula in an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery presenting as an acute coronary syndrome&#46; To the best of our knowledge&#44; this is the first case in the literature involving a coronary artery fistula in an intercoronary communication&#46;</p>"
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        "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A comunica&#231;&#227;o intercoron&#225;ria &#233; uma anomalia muito rara da art&#233;ria coron&#225;ria&#46; Define-se como circula&#231;&#227;o em aberto com um fluxo sangu&#237;neo bidirecional entre as duas art&#233;rias coron&#225;rias&#46; As f&#237;stulas das art&#233;rias coron&#225;rias s&#227;o comunica&#231;&#245;es anormais entre a art&#233;ria coron&#225;ria e uma c&#226;mara card&#237;aca ou um vaso principal&#46; Um homem de 62 anos foi admitido no nosso hospital revelando fraqueza geral s&#250;bita&#44; tonturas e sensa&#231;&#227;o de compress&#227;o no peito&#46; No momento apresentava press&#227;o arterial de 80 mmHg&#47;40 mmHg e frequ&#234;ncia card&#237;aca de 65 batimentos&#47;min&#46; O eletrocardiograma &#40;ECG&#41; revelou ritmo sinusal e eleva&#231;&#227;o do segmento ST de 1-2 mm nas deriva&#231;&#245;es anteriores&#46; O doente foi conduzido para o laborat&#243;rio de hemodin&#226;mica para interven&#231;&#227;o coron&#225;ria percut&#226;nea&#46; O tronco comum e as art&#233;rias coron&#225;rias circunflexas esquerdas estavam normais&#46; A angiografia coron&#225;ria mostrou uma comunica&#231;&#227;o entre o tronco comum e o ramo diagonal da art&#233;ria descendente anterior e uma f&#237;stula entre a comunica&#231;&#227;o intercoron&#225;ria e a aur&#237;cula esquerda&#46; As outras art&#233;rias coron&#225;rias estavam normais&#46; Os resultados dos testes laborat&#243;riais&#44; incluindo os n&#237;veis de troponina I card&#237;aca e de creatina-kinase MB estavam normais&#46; Os sintomas de angina desapareceram e a eleva&#231;&#227;o do segmento ST foi resolvida ap&#243;s 4 horas&#46; Relatamos um caso interessante de uma f&#237;stula cong&#233;nita da art&#233;ria coron&#225;ria na comunica&#231;&#227;o intercoron&#225;ria entre o tronco comum e o ramo diagonal da art&#233;ria coron&#225;ria descendente anterior esquerda apresentadas como uma s&#237;ndrome coron&#225;ria aguda&#46; De acordo com o nosso conhecimento relatamos o primeiro caso na literatura que envolve uma f&#237;stula da art&#233;ria coron&#225;ria na comunica&#231;&#227;o intercoron&#225;ria&#46;</p>"
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Vol. 33. Issue 1.
Pages 55.e1-55.e4 (January 2014)
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Vol. 33. Issue 1.
Pages 55.e1-55.e4 (January 2014)
Case report
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Congenital coronary artery fistula in an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery: An interesting case report
Fístula congénita da artéria coronária na comunicação intercoronária entre o trono comum e um ramo diagonal da artéria coronária descendente anterior esquerda: um caso clínico interessante
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Yasin Turker
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dryasinturker@hotmail.com

Corresponding author.
, Hakan Tibilli
Department of Cardiology, Duzce University, Duzce, Turkey
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Abstract

Intercoronary communication is a very rare coronary artery anomaly. It is defined as an open-ended circulation with bidirectional blood flow between two coronary arteries. Coronary artery fistulas are abnormal communications between a coronary artery and a cardiac chamber or major vessel. A 62-year-old man was admitted to our hospital with sudden development of general weakness, dizziness and a sensation of compression in his chest. At presentation his blood pressure was 80/40 mmHg and heart rate was 65 beats/min. The ECG revealed sinus rhythm and 1–2 mm ST elevation in the anterior leads. The patient was taken to the catheterization laboratory for percutaneous coronary intervention. The left main and left circumflex coronary arteries were normal. Coronary angiography showed a communication between the left main and the diagonal branch of the left anterior descending and a fistula between the intercoronary connection and the left atrium. The other coronary arteries were normal. Laboratory test results, including cardiac troponin I and creatine kinase–MB levels, were normal. The angina symptoms disappeared and the ST elevation resolved within four hours. We report an interesting case of congenital coronary artery fistula in an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery presenting as an acute coronary syndrome. To the best of our knowledge, this is the first case in the literature involving a coronary artery fistula in an intercoronary communication.

Keywords:
Acute coronary syndrome
Coronary artery fistula
Intercoronary communication
Resumo

A comunicação intercoronária é uma anomalia muito rara da artéria coronária. Define-se como circulação em aberto com um fluxo sanguíneo bidirecional entre as duas artérias coronárias. As fístulas das artérias coronárias são comunicações anormais entre a artéria coronária e uma câmara cardíaca ou um vaso principal. Um homem de 62 anos foi admitido no nosso hospital revelando fraqueza geral súbita, tonturas e sensação de compressão no peito. No momento apresentava pressão arterial de 80 mmHg/40 mmHg e frequência cardíaca de 65 batimentos/min. O eletrocardiograma (ECG) revelou ritmo sinusal e elevação do segmento ST de 1-2 mm nas derivações anteriores. O doente foi conduzido para o laboratório de hemodinâmica para intervenção coronária percutânea. O tronco comum e as artérias coronárias circunflexas esquerdas estavam normais. A angiografia coronária mostrou uma comunicação entre o tronco comum e o ramo diagonal da artéria descendente anterior e uma fístula entre a comunicação intercoronária e a aurícula esquerda. As outras artérias coronárias estavam normais. Os resultados dos testes laboratóriais, incluindo os níveis de troponina I cardíaca e de creatina-kinase MB estavam normais. Os sintomas de angina desapareceram e a elevação do segmento ST foi resolvida após 4 horas. Relatamos um caso interessante de uma fístula congénita da artéria coronária na comunicação intercoronária entre o tronco comum e o ramo diagonal da artéria coronária descendente anterior esquerda apresentadas como uma síndrome coronária aguda. De acordo com o nosso conhecimento relatamos o primeiro caso na literatura que envolve uma fístula da artéria coronária na comunicação intercoronária.

Palavras-chave:
Síndrome coronária aguda
Fístula da artéria coronária
Comunicação intercoronária
Full Text
Introduction

Intercoronary communication is a very rare coronary artery anomaly, with a prevalence of 2.37/100 000. It is defined as an open-ended circulation with bidirectional blood flow between two coronary arteries.1 It can be distinguished from collateral arteries by its angiographic features, and in itself does not usually reflect underlying coronary artery disease.2 Intercoronary arterial connections are thought to be congenital in origin. Compared with collaterals, intercoronary arterial connections are larger in diameter (>1 mm), extramural, and straight. Furthermore, the structure of an intercoronary arterial connection is typical of an epicardial coronary artery, with a well-defined muscular layer.3

Coronary artery fistulas are abnormal communications between a coronary artery and a cardiac chamber or major vessel.4 They may be congenital or acquired due to trauma or iatrogenic causes. Angiographic series reveal an incidence of coronary artery fistula in adults of 0.3–0.8%.4–6 Most of these patients are asymptomatic, but heart failure, angina, myocardial infarction, coronary steal, endocarditis, and dyspnea have been reported.7

We report an interesting case of congenital coronary artery fistula in an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery presenting as an acute coronary syndrome.

To the best of our knowledge, this is the first case in the literature involving a coronary artery fistula in an intercoronary communication.

Case report

A 62-year-old man was admitted to our hospital with sudden development of general weakness, dizziness and a sensation of compression in his chest. He had a history of diabetes mellitus. At presentation his blood pressure was 80/40 mmHg and heart rate was 65 beats/min. The ECG revealed sinus rhythm and 1–2 mm ST elevation in the anterior leads (Figure 1). The patient was taken to the catheterization laboratory for percutaneous coronary intervention. The left main and left circumflex coronary arteries were normal. Coronary angiography showed a communication between the left main and the diagonal branch of the left anterior descending and a fistula between the intercoronary connection and the left atrium (Figure 2). The other coronary arteries were normal. Laboratory test results, including cardiac troponin I and creatine kinase–MB levels, were normal. The angina symptoms disappeared and the ST elevation resolved within four hours (Figure 3). To the best of our knowledge, this is the first case in the literature involving a congenital coronary artery fistula in an intercoronary communication.

Figure 1.

Electrocardiogram revealing 1–2 mm ST elevation in the anterior leads.

(0.72MB).
Figure 2.

Contrast injection in the left coronary artery showing a large connection between the left main and the diagonal branch of the left anterior descending (black arrow) and a fistula between the intercoronary connection and the left atrium (white arrow). D: diagonal; LM: left main.

(0.16MB).
Figure 3.

Electrocardiogram demonstrating normal sinus rhythm.

(0.29MB).
Discussion

The hemodynamic consequences of coronary artery fistulas are variable depending on shunt size, site of the shunt and presence of other underlying heart disease.8 Fifty percent of patients with large or multiple fistulas may develop complications, which may include bacterial endocarditis, thrombosis, aneurysm formation, dissection, rupture, premature atherosclerosis, pulmonary hypertension, myocardial ischemia, or infarction.9

The functional significance of large intercoronary communications between normal coronary arteries is unclear but one may speculate that they have a potential role in protecting the myocardium should significant atherosclerosis develop in either of the patent arteries3; on the other hand, myocardial ischemia could result if an unidirectional intercoronary communication causes a coronary steal phenomenon that results in inadequate perfusion.10 The ischemic consequences of an intercoronary connection with unidirectional flow may be explained by its similarity to a fistula from a coronary artery to a low-pressure cardiac space.11

We believe that an intercoronary communication between the left main and the diagonal branch of the left anterior descending coronary artery and a congenital coronary artery fistula may be the cause of ischemia in this case.

Ethical disclosuresProtection of human and animal subjects

The authors declare that the procedures followed were in accordance with the regulations of the relevant clinical research ethics committee and with those of the Code of Ethics of the World Medical Association (Declaration of Helsinki).

Confidentiality of data

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.

Right to privacy and informed consent

The authors have obtained the written informed consent of the patients or subjects mentioned in the article. The corresponding author is in possession of this document.

Conflicts of interest

The authors have no conflicts of interest to declare.

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Coronary artery fistulae.
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Arteriovenous connection between the aorta and the coronary sinus through a giant fistulous right coronary artery.
Hellenic J Cardiol, 49 (2008), pp. 48-51
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