Anomalous Origin of Coronary Arteries

Rev Bras Cardiol Invas 2007; 15(3).Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).Anomalous Origin of Coronary ArteriesReview ArticleFlavio Henrique Amaral Pires Veras 1 , Edgar Guimarães Victor 1 ,Lurildo Cleano Ribeiro Saraiva 1 , Marly Maria Uellendahl Lopes 1SUMMARYThe anomalous origin of the coronary arteries is a potentiallylethal pathology, especially in neonates and young athletes.The understanding of the pathophysiological pattern of thedisease has allowed the recognition of the different formsof clinical presentation. Despite being a cause of myocardialischemia, functional tests can be normal. Coronaryangiography used to be the gold standard for the diagnosisof this pathology, but non-invasive cardiovascular imagingtests have shown better definition of the origin and courseof coronary arteries. Surgical treatment should be indicatedfor symptomatic patients, mainly in children with theanomalous origin of the left coronary artery from thepulmonary artery. The ideal management of asymptomaticpatients remains undefined.DESCRIPTORS: Coronary vessel anomalies. Death, sudden.Diagnostic imaging. Diagnostic techniques, cardiovascular.RESUMOOrigem Anômala das Artérias CoronáriasA origem anômala das artérias coronárias é uma doençapotencialmente letal, sobretudo em lactentes e atletasjovens. O entendimento da fisiopatologia da doença permitiua diferenciação entre as diferentes apresentaçõesclínicas. Apesar de ser causa de isquemia miocárdica, osexames indutores de isquemia podem ser normais. Acineangiocoronariografia foi durante muito tempo consideradao exame ideal para o diagnóstico da doença, masos exames de imagem cardiovascular não-invasivos têmmostrado uma melhor definição da origem e do trajetodas artérias coronárias. O tratamento cirúrgico deve serindicado em pacientes sintomáticos, principalmente emcrianças com origem anômala da artéria coronária esquerdada artéria pulmonar. O manejo ideal para ospacientes assintomáticos permanece indefinido.DESCRITORES: Anomalias dos vasos coronários. Mortesúbita. Diagnóstico por imagem. Técnicas de diagnósticocardiovascular.The congenital anomaly of coronary arteries canbe benign or potentially serious, causing myocardialischemia, infarct and sudden death 1 . The incidencevaries between 0.3% to 1.5% in necropsy studies orthrough cineangiocoronography (CA) 2-7 . Angelini et al. 8 ,in a study which included 1950 consecutive CA’s forthe assessment of coronary arterial illness, found anincidence of 5.6%.The origin and the proximal course of anomalouscoronary arteries are the main factors that predict theseriousness of the illness 1 . Currently, they constitute thesecond most frequent cause of sudden death ofcardiovascular origin in competitive athletes 9 . It can alsobe cause of heart insufficiency and death in babies 10,11 .1Hospital das Clínicas da Universidade Federal de Pernambuco,Recife, PE.Correspondence: Flavio Henrique A. P. Veras. Serviço de Cardiologiado Hospital das Clínicas da UFPE. Av. Prof. Moraes Rego, SN -Cidade Universitária - Recife, PE - CEP: 50670-240Phone: (81) 2126-3714 - E-mail: fhveras@bol.com.brRecepted in: 6/5/2007 • Acept in: 29/7/2007The diagnosis of this anomaly is difficult sincethe subject might be asymptomatic until the momentof the lethal event 1,12 . Complementary exams, especiallythose of cardiovascular imaging, are important toolsfor diagnosis.In the anomalous origin of the left coronary arteryof the pulmonary artery (OACEP), surgical treatmenthas excellent results 10,13 . The therapeutics of the anomalousorigin of the coronary arteries of the aorta(OACEA) are an object of constant discussion.CLASSIFICATION AND DEFFINITIONThe first classification for coronary artery anomalieswas proposed by Ogden 14 , in 1969. This classificationsubdivided coronary anomalies into major, minor andsecondary, anatomically based, but did not propose aclinical correlation 15 .Dodge-Khatami et al. 15 subdivided coronaryanomalies into seven categories, according to clinicalcomplexity criteria. These would be: coronary arteriesoriginating from the pulmonary artery, coronary arteries1

Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).with an anomalous origin in the aorta, congenital atresiaof the left coronary trunk, coronary arteriovenous fistulas,coronary arteries forming myocardial bridges, aneurysmsof coronary arteries and coronary stenosis.For Angelini et al. 8 , a coronary anomaly should bedefined as any coronary pattern, with a characteristic(number of ostios, proximal course, distal bed… ) “rarely”encountered in the general population. The followingdefinitions being favorable: normal, any morphologicalcharacteristic observed in more than 1% of a nonselectedpopulation; variable of normality, a relativelyuncommon morphological characteristic prevalent inmore than 1% of the population; and anomaly, a morphologicalcharacteristic seen in less than 1% of thatpopulation. Normal coronary anatomy, universally accepted,is defined in this way 16,17 :• The coronary trunk originates from the left coronarysinus and divides into descendent anterior arteryand circumflex artery;• The anterior descendent artery follows behindthe pulmonary trunk in the intraventricular anteriorgrove;• The circumflex artery follows the posterioratrioventricular grove;• The right coronary artery originates from the rightcoronary sinus and follows along the anterioratrioventricular grove.Any alteration in this pattern must be consideredas an anomalous coronary anatomy 16,17 . The circumflexcoronary artery, originating from the right coronarysinus or from the right coronary artery, with a retroaorticcourse, is the most common congenital anomalyof the coronary artery 18, 19 .PHYSIOPATHOLOGYCoronary anomalies have been implicated as a causefor thoracic pain, sudden death, heart insufficiency,syncope, dyspnea, ventricular fibrillation and AMI 8 .In the OACEP, it is observed that, during intrauterinelife, no functional alteration takes place, however,after birth, in the measure that arteriolar resistance fallsand blood pressure decreases in the pulmonary artery,the oxi-hemoglobinic saturation of the venous blooddiminishes, the arterial channel and the oval foramenclose, progressively diminishing the myocardial bloodperfusion, irrigated by the anomalous artery, leadingto ischemic alterations.The OACEP is generally an isolated defect, butmight be associated, in about 5% of cases, to othercardiac defects such as, intraventricular communication,interatrial communication, aortal coarctation, Fallot’sTetralogy and other anomalies 13 . In Fallot’s Tetralogybearers, the incidence of coronary anomaly can varyfrom 3% to 36% 20,21 .The congenital atresy of the left coronary trunk(LCT) is an extremely rare anomaly and generally hasa benign evolution 22 . In some cases, it can lead tomyocardium ischemia and sudden death 22 . The bearersof this anomaly survive thanks to the development ofcollateral circulation for the anterior descendent artery 22 .Collateral circulation can be made up by branches/bunches from the cone artery, also known as VieussensCircle; or anastomosis between the anterior ventricularbranches of the right coronary artery and the anteriordescendent artery 22,23 .The explanation for myocardial ischemia in theatresia of the TCE, in the absence of arterioscleroses orvessel spasm, is not clear and the most logical justificationis the fact that an only coronary artery is incapable ofsupplying, adequately, the myocardial blood demand 22 .The anomalous origin of the left coronary arteryof the right coronary sinus can be related to suddendeath in 59% of cases, preceded by physical activityin 81% of events 8 . This anomaly can present 4 courses:anterior, to the pulmonary artery; posterior, to the aorta;intra-septal, between the aorta and the pulmonary artery;and intra-arterial, between the aorta and the pulmonary(Figure 1) 24 . All the subtypes of this anomaly have beenassociated to sudden death, but the intra-arterial variety,which is the most common pattern, has a strongerrelation with catastrophic events 1,8,24-26 .Potential mechanisms have been erected to explainthe presence of myocardial ischemia and sudden deathin patients with OACEA; the formation of an acute andcontorted angle at the origin of the anomalous coronaryof the aorta; a narrowing of the coronary orifice, secondaryto the anomalous anatomy; compression ofthe anomalous coronary artery during its course betweenthe aorta and the pulmonary trunk during exercise; andspasm of the anomalous artery, possibly as a result ofendothelial injury 8,27,28 .According with current evidence, the coronarysegments with anomalous course are not more susceptibleto obstructive arterosclerotic disease than normalsegments in a same individual 8,9 (Figure 1).CLINICAL OUTLOOKThe OACEP presents two levels: an infantile formand an adult form. The infantile form is characterizedby poor intercoronary collateral circulation, which canresult in AMI, cardiac insufficiency or sudden death 11 .In this type of presentation, the symptoms appear in thefirst months of life, with constant crying, intense paleness,sudden stop when suckling, weight loss and signs ofcardiac insufficiency, such as taquipnea tachycardia,tiredness while suckling, sweating, hepathomegalia andperipherical cyanosis. On auscultation, a galloping rhythmcan be heard, muffling of the bustle, especially thefirst one, and occasionally, the appearance of systolic2

Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).Figura 1 - Origem anômala da artéria coronária esquerda no seio de Valsalva direito. Cada painel tem uma representação de um cortecraniocaudal, ao nível das válvulas semilunares, mostrando o trajeto da artéria coronária anômala. Os angiogramas em posição oblíqua anteriordireita e as representações esquemáticas mostram exemplos dos quatro trajetos mais comuns de artéria coronária esquerda anômala originandosedo seio de Valsalva: posterior (retroaórtico), interarterial, anterior e septal. CD: artéria coronária direita, DA: artéria descendente anterior;Cx: artéria circunflexa (adaptado de Popma et al. 27 ).3

Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).murmur in the mitral area, as a result of secondarymitral insufficiency to the increase of the cardiac areadue to its dilatation or due to dysfunction of the papillarymuscle 10,11 . Mortality during the first year of life isestimated at 90%, even with clinical treatment 10,11 .The adult or mature form can occur in childrenand necessarily in adults in around 10 – 15% of cases.It is characterized by an abundance of intercoronarycollateral circulation, of sufficient magnitude to allowa survival rate until adult age, in some cases, it hasbeen told, until 72 years of age.Sometimes, there are no appreciable clinical manifestationsand the diagnosis of OACEP is suspecteddue to the presence of murmurs: continuous, as aresult of the passage of blood through the coronarypulmonaryfistula, or systolic, because of dysfunctionof the mitral valve 10 . Some patients evolve towardscardiac insufficiency; others towards angina 10,11 .In the OACEA, some symptoms such as syncope,chest pain and palpitations may be present 12,28 . Butmost bearers are asymptomatic and the only expressionof this coronary anomaly in young athletes might besudden death 12,28 . The studies suggest that these coronaryanomalies can be lethal only during or immediatelyafter extenuating physical exercise, typically in youngsubjects 8 . (Table 1).COMPLEMENTARY EXAMSElectrocardiogram (ECG) and ergometric test (ET)One study demonstrated that the presence of the“Q” wave with a depth over 3 mm, duration higherthan 30 ms and a “QR” pattern in at least one of thedeviations – DI, aVL, V5-V7, was present in 100% ofexams of patients with OACEP 29 . The absence of the“Q” wave in the lower wall is also a prevalent finding(82% of cases) 13,15,29 . The axel deviation towards theleft is less frequent (64% of cases) 13 . Atypical electrocardiographicalterations can mask the illness whenassociated to lesions with important pulmonary hipoflux.In the OACEA, the ECG and the TE are almostalways normal 8,9,12,28 . In spite of the OACEA being thecause of myocardial ischemia, this is incidental, with manifestationsunder extenuating clinical conditions 8,9,12,28 .Thorax X-RayIn general, it shows alterations in the cases associatedto cardiac insufficiency. In the OACEP, young agedchildren can present an increase of the cardiac area 10 .Lung circulation is normal, however, when cardiacinsufficiency is present, a picture of venous pulmonarycongestion can be observed 10 .EchocardiogramThe bidimensional echocardiogram with coloredDoppler is useful in the morphofunctional assessmentof the heart. For the diagnosis of anomalous origin ofthe coronary arteries, the studies show a variablesensibility, with better results in children 11,13,15,26 .The transesophageal echocardiogram (TEE) is anexam with a much higher sensibility for the detectionof the anomalous origin of coronary arteries 1,26 . TheTEE, specially with the use of a multi-plane probe/catheter, is capable of drawing, with precision, theproximal course and the flux pattern in the anomalouscoronary arteries 1,26 .Computer Tomography of Coronary Arteries (CTCA)The studies show a quick evolution of this noninvasivemethod in the assessment of coronary arteries.Initially, the studies with the Electron Beam ComputedTomography (EBCT), demonstrated excellent accuracyin the assessment of the origin and proximal course ofcoronary anomalies 19,25 .With the advent of Computer Tomography withMultiple Detectors (16 and 64 acquisitions), a betterassessment of the middle and distal segments waspossible, for the detection of obstructive coronary arteryillness 16. It uses ionizing radiation and requires theuse of medication to diminish heart frequency 25. Themethod requires the administration of a contrast meansin the veins, which is potentially allergic and nephrotoxic.The data in the literature show it to be a safe andefficient method with excellent spatial resolution 16,24,25,30 .Comparative studies with coronary angiography haveshown a higher efficacy, suggesting that ComputerTomography with Multiple Detectors is the ideal examfor the detection of coronary anomalies 16,19 .TABELA 1Incidência de morte súbita relacionada a anomalias de artérias coronárias (adaptado de Angelini et al. 8 ).Grupo (idade) Número de mortes Mortes relacionadas a anomalias coronarianas,%Indivíduos fazendo exercício (8-66 a) 550 11População geral (< 40 a) 162 0,6Atletas competitivos(idade média de 17 a) 134 23Corredores (30-46 a) 120 1,6Indivíduos fazendo exercício, Maryland - EUA 62 04

Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).Figure 2 illustrates a CTCA where an OACEA withan inter-arterial course is evidenced.Nuclear Magnetic Resonance (NMR)of the coronary arteriesCoronary angiography through MR is a noninvasiveexam, with high accuracy for the anatomicalidentification of the origin and proximal course of thecoronary arteries. The free choice of the image plane,with a trydimensional view, is an important advantageover the limited angle possibilities of conventionalcoronary angiography. It requires a non-nephrotoxiccontrast and does not use ionizing radiation 16 .It has the disadvantage of the long time of theexam (45-50 minutes), the need of periods of apnea,and the limitation of the analysis of the distal coronarysegments 19,31-34 .Bunce et al. 34 developed a technique for the detectionof coronary anomalies were there is no needof apnea and with a shorter time of exam (average time26 minutes).It is an exam that possibilitates cardiac morphofunctionalassessment, being of great usefulness in congenitalcardiopathies, since it allows a perfect spatialassessment between the coronary arteries, the greatvessels and all other cardiac structures 19,31-34 .Figure 3 illustrates a NMR diagnosing an athresiaof the TCE.CineangiocoronarographyIt is traditionally considered the ideal exam for thediagnosis of coronary anomalies. It is an invasive exam,which uses nephro-toxic contrast and ionizing radiation.The evolution of the RNM and of the TCAC, bothwith trydimensional analysis, have shown failures inthe CA for the diagnosis of coronary anomalies 19,31-33 .The CA might be incapable of differentiating, duringthe course through an anomalous coronary, the highrisk intra-arterial anatomy, from the septal anatomyretro-aortal or of the anterior wall, as well as the identificationof the coronary sinus and the morphology ofthe coronary ostium in the wall of the aorta 19,31-33 .The CA is still the best method for the assessmentof the distal coronary bed and associated arteroscleroticlesions 19,35,36 .TREATMENTOnce diagnosed, the OACEP must be surgicallycorrected immediately after the diagnosis, to avoidcomplications and sequels, characteristic of the naturalhistory of this illness 10,11,13,37-40 .The chosen surgical technique is the reimplantationwith translocation of the left coronary artery of thepulmonary trunk to the aorta, which is possible inmost cases 11,13,37-40 .In the case in which this technique is impossible toapply, “tunnelization” or Takeuchi Technique is recommended,which consists of creating a tunnel inside thepulmonary artery, to connect the left coronary arterywith the aorta 11,13,37-40 . Other surgical options are simplyto string or thread, to tie the left coronary artery with theaorta 11,13,37-40 . Surgical mortality for all the combinedFigura 2 - Origem anômala da artéria coronária esquerda com curso interarterial visualizada por TCAC. A: corte axial para avaliação da origemdas artérias coronárias. B: reconstrução tridimensional. As setas indicam o trajeto da artéria descendente anterior entre a aorta ascendentee o tronco da artéria pulmonar. A cabeça da seta indica a artéria coronária esquerda. Ao: aorta; AP: artéria pulmonar; AD: átrio direito; AE:átrio esquerdo(adaptado de Ropers et al. 24 ).5

Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).techniques varied between 75 and 80% in the 80s 37 .Nowadays, mortality is situated between 0 and 23% 37 .The clinical evolution and functional recovery ofthe left ventricle will depend upon the dysfunctionalventricular degree before the surgery. In most cases,there is a normalization of the ventricular function afterthe operation 11,13,37-40 .Bearers of OACEA, in general, are asymptomatic 12 .Even in athletes, only a minority is symptomatic 12 . And,in symptomatic individuals, the ECG and ischemia inducingexams, are generally normal 8,12,28 . The presenceof symptoms suggestive of the illness, especially inathletes, must be investigated through imaging exams.In the case in which echocardiograms are unable todiagnose the anomalous origin, TCAC, RNM or CA ofthe coronary arteries must be performed 8,28 .There is no consensus in the literature for the managementof asymptomatic bearers of OACEA. In symptomaticpatients, the surgical option must be chosen 12,28 .Figura 3 - Atresia do TCE em um paciente portador de valva aórtica bicúspide e ducto arterioso patente. A: cineangiocoronariografia mostrandoque a artéria circunflexa(Cx) origina-se da artéria coronária direita(CD). A artéria descendente anterior(DA) enche-se retrogradamente e formauma fístula com a artéria pulmonar(AP). B: no corte axial da RNM, observa-se uma falsa inserção da DA na AP. C: imagem axial da Cxoriginando-se da CD. Ao: aorta; AD: átrio direito; VE: ventrículo esquerdo(adaptado de Taylor et al. 17 )6

Veras FHAP, et al. Anomalous Origin of Coronary Arteries. Rev Bras Cardiol Invas 2007; 15(3).The surgical options are the implant of arterial or venousgraft or, reimplantation of the anomalous coronary arteryin the anatomical coronary sinus, plastia of the intramuralsegment of the coronary artery and translocation of thepulmonary artery trunk 12,28 . There is no data which definesthe best surgical technique.CONCLUSIONThe anomalous origin of coronary arteries is arare illness, but potentially lethal if not diagnosed andtreated early. Children and young athletes are the riskgroup for the catastrophic sequelae. In these patients,therapeutic surgery brings good results, being this adefinitive treatment. The identification of the illness inasymptomatic patients continues being a challenge.And, new studies are necessary, for the definition ofthe ideal treatment in these patients.BIBLIOGRAPHIC REFERENCES1. Dawn B, Talley JD, Prince CR, Hoque A, Morris GT,Xenopoulos NP, et al. 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