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

RevPortCardiol.2014;33(1):55.e1---55.e4

Revista

Portuguesa

de

Cardiologia

Portuguese

Journal

of

Cardiology

www.revportcardiol.org

CASE

REPORT

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

Yasin

Turker

,

Hakan

Tibilli

DepartmentofCardiology,DuzceUniversity,Duzce,Turkey

Received17June2013;accepted4July2013 Availableonline9January2014

KEYWORDS Acutecoronary syndrome; Coronaryartery fistula; Intercoronary communication

Abstract Intercoronarycommunicationisaveryrarecoronaryarteryanomaly.Itisdefinedas anopen-endedcirculationwithbidirectionalbloodflowbetweentwocoronaryarteries. Coro-nary arteryfistulasareabnormal communications betweenacoronaryartery andacardiac chamberormajorvessel.A62-year-oldmanwasadmittedtoourhospitalwithsudden develop-mentofgeneralweakness,dizzinessandasensationofcompressioninhischest.Atpresentation hisbloodpressurewas80/40mmHgandheartratewas65beats/min.TheECGrevealedsinus rhythmand1---2mmSTelevationintheanteriorleads.Thepatientwastakentothe catheter-izationlaboratoryforpercutaneouscoronaryintervention.Theleft mainandleftcircumflex coronaryarterieswerenormal.Coronaryangiographyshowedacommunicationbetweenthe left mainandthediagonalbranchoftheleft anteriordescendingandafistulabetweenthe intercoronaryconnectionandtheleftatrium.Theothercoronaryarterieswerenormal. Lab-oratorytestresults,includingcardiactroponinIandcreatinekinase---MBlevels,werenormal. TheanginasymptomsdisappearedandtheSTelevationresolvedwithinfourhours.Wereport aninteresting case ofcongenitalcoronaryartery fistulainanintercoronarycommunication betweentheleftmainandthediagonalbranchoftheleftanteriordescendingcoronaryartery presentingasanacutecoronarysyndrome.Tothebestofourknowledge,thisisthefirstcase intheliteratureinvolvingacoronaryarteryfistulainanintercoronarycommunication. © 2013 Sociedade Portuguesa de Cardiologia Published by Elsevier España, S.L. All rights reserved.

∗_{Correspondingauthor.}

E-mailaddress:dryasinturker@hotmail.com(Y.Turker).

0870-2551/$–seefrontmatter©2013SociedadePortuguesadeCardiologiaPublishedbyElsevierEspaña,S.L.Allrightsreserved.

55.e2 Y.Turker,H.Tibilli PALAVRAS-CHAVE Síndromecoronária aguda; Fístuladaartéria coronária; Comunicac¸ão intercoronária

Fístulacongénitadaartériacoronárianacomunicac¸ãointercoronáriaentreotrono comumeumramodiagonaldaartériacoronáriadescendenteanterioresquerda:um casoclínicointeressante

Resumo A comunicac¸ão intercoronária é uma anomalia muito rara da artéria coronária. Define-secomocirculac¸ãoemabertocomumfluxosanguíneobidirecionalentreasduasartérias coronárias.Asfístulasdasartériascoronáriassãocomunicac¸õesanormaisentreaartéria coro-náriaeuma câmaracardíacaouum vasoprincipal.Umhomemde 62anosfoi admitidono nossohospitalrevelandofraquezageralsúbita,tonturasesensac¸ãodecompressãonopeito. Nomomentoapresentavapressãoarterialde80mmHg/40mmHgefrequênciacardíacade65 batimentos/min.Oeletrocardiograma(ECG)revelouritmosinusaleelevac¸ãodosegmentoSTde 1-2mmnasderivac¸õesanteriores.Odoentefoiconduzidoparaolaboratóriodehemodinâmica paraintervenc¸ãocoronáriapercutânea.Otroncocomumeasartériascoronáriascircunflexas esquerdasestavamnormais.Aangiografiacoronáriamostrouumacomunicac¸ãoentreotronco comumeoramodiagonaldaartériadescendenteanterioreumafístulaentreacomunicac¸ão intercoronáriaeaaurículaesquerda.Asoutrasartériascoronáriasestavamnormais.Os result-adosdostesteslaboratóriais,incluindoosníveisdetroponinaIcardíacaedecreatina-kinase MBestavamnormais.Ossintomasdeanginadesaparecerameaelevac¸ãodosegmentoSTfoi resolvidaapós4horas.Relatamos umcasointeressantede umafístulacongénitadaartéria coronárianacomunicac¸ãointercoronáriaentreotroncocomumeoramodiagonaldaartéria coronáriadescendenteanterioresquerdaapresentadascomoumasíndromecoronáriaaguda. Deacordocomonossoconhecimentorelatamosoprimeirocasonaliteraturaqueenvolveuma fístuladaartériacoronárianacomunicac¸ãointercoronária.

© 2013 SociedadePortuguesa de Cardiologia. Publicado por Elsevier España, S.L.Todos os direitosreservados.

Introduction

Intercoronarycommunicationisaveryrarecoronaryartery anomaly,withaprevalence of 2.37/100 000.It isdefined asanopen-endedcirculation withbidirectionalbloodflow between two coronary arteries.1 _{It can be distinguished}

fromcollateral arteries by itsangiographic features, and initselfdoesnotusuallyreflectunderlyingcoronaryartery disease.2_{Intercoronaryarterialconnectionsarethoughtto}

becongenital in origin. Compared withcollaterals, inter-coronary arterial connections are larger in diameter (>1 mm), extramural, and straight. Furthermore, the struc-ture of an intercoronary arterial connection is typical of anepicardialcoronaryartery,withawell-definedmuscular layer.3

Coronary artery fistulas are abnormal communications betweenacoronaryarteryandacardiacchamberormajor vessel.4_{Theymaybecongenitaloracquiredduetotrauma}

oriatrogeniccauses.Angiographicseriesrevealanincidence ofcoronaryarteryfistulain adultsof 0.3---0.8%.4---6_Mostof

thesepatientsareasymptomatic,butheartfailure,angina, myocardialinfarction,coronarysteal,endocarditis,and dys-pneahavebeenreported.7

We report an interesting case of congenital coronary arteryfistulain an intercoronarycommunication between theleftmain andthediagonalbranchof theleftanterior descendingcoronaryarterypresentingasanacutecoronary syndrome.

Tothebestofourknowledge,thisisthefirstcaseinthe literatureinvolvingacoronaryarteryfistulainan intercoro-narycommunication.

Case

report

A62-year-oldmanwasadmittedtoourhospitalwithsudden developmentofgeneralweakness,dizzinessandasensation ofcompressioninhischest.Hehadahistoryofdiabetes mel-litus.At presentationhisblood pressurewas80/40 mmHg andheart ratewas65beats/min.The ECG revealedsinus rhythm and 1---2 mm ST elevation in the anterior leads (Figure 1). The patient wastaken to the catheterization laboratoryforpercutaneouscoronaryintervention.Theleft main and left circumflex coronary arteries were normal. Coronary angiography showed a communication between the left main and the diagonal branch of the left ante-rior descending and a fistula between the intercoronary connectionandtheleftatrium(Figure2).Theother coro-naryarterieswerenormal.Laboratorytestresults,including cardiactroponinIandcreatinekinase---MBlevels,were nor-mal.TheanginasymptomsdisappearedandtheSTelevation resolved within four hours (Figure 3). To the best of our knowledge, this is the first case in the literature involv-ingacongenitalcoronaryarteryfistulainanintercoronary communication.

Discussion

The hemodynamic consequences of coronary artery fis-tulas 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

Congenitalcoronaryarteryfistula 55.e3 10 mm/mV 25 mm/s Filter: H50 d 100 Hz I V1 V₂ V3 V4 V5 V6 II III aVR aVL aVF 5 mm/mV

Figure1 Electrocardiogramrevealing1---2mmSTelevationintheanteriorleads.

D

LM

Figure2 Contrastinjectionintheleftcoronaryartery show-ingalargeconnectionbetweentheleftmainandthediagonal branchoftheleftanteriordescending(blackarrow)anda fis-tulabetweentheintercoronaryconnectionandtheleftatrium (whitearrow).D:diagonal;LM:leftmain.

endocarditis,thrombosis,aneurysmformation, dissection, rupture, premature atherosclerosis, pulmonary hyperten-sion,myocardialischemia,orinfarction.9

The functional significance of large intercoronary communications between normal coronary arteries is unclear but one may speculate that they have a poten-tial role in protecting the myocardium should significant atherosclerosisdevelopineitherofthepatentarteries3_;on

theotherhand,myocardialischemiacouldresultifan uni-directionalintercoronarycommunicationcausesacoronary steal phenomenon that results in inadequate perfusion.10

Theischemicconsequencesofanintercoronaryconnection withunidirectionalflowmaybeexplainedbyitssimilarity toafistulafromacoronaryarterytoalow-pressurecardiac space.11

We believe that an intercoronary communication betweentheleftmainandthediagonalbranchoftheleft anteriordescendingcoronaryarteryandacongenital coro-naryarteryfistulamaybethecauseofischemiainthiscase.

Ethical

disclosures

Protection of human and animal subjects.The authors declarethat the proceduresfollowed were inaccordance withtheregulationsoftherelevantclinicalresearchethics

I _V 1 V₂ V3 V₄ V5 V₆ II III aVR aVL aVF

55.e4 Y.Turker,H.Tibilli committeeandwiththoseoftheCodeofEthicsoftheWorld

MedicalAssociation(DeclarationofHelsinki).

Confidentialityofdata.Theauthorsdeclarethattheyhave followedtheprotocolsoftheirworkcenteronthe publica-tionofpatientdataandthatallthepatientsincludedinthe studyreceivedsufficientinformationandgavetheirwritten informedconsenttoparticipateinthestudy.

Righttoprivacyandinformedconsent.Theauthorshave obtainedthe written informed consentof the patients or subjectsmentionedinthearticle.Thecorrespondingauthor isinpossessionofthisdocument.

Conflicts

of

interest

Theauthorshavenoconflictsofinteresttodeclare.

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