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Ann Thorac Surg 1998;66:1444-1449
© 1998 The Society of Thoracic Surgeons

Current Review

Anomalous origin of the right coronary artery: preoperative and postoperative hemodynamics

Peter W. Radke, MDa, Bruno J. Messmer, MDb, Philipp K. Haager, MDa, Heinrich G. Klues, MDa

a Medical Clinic I, RWTH University Hospital, Aachen, Germany
b Department of Cardiothoracic Surgery, RWTH University Hospital, Aachen, Germany

Address reprint requests to Dr Klues, Medical Clinic I, RWTH University Hospital, Pauwelsstraße 30, D-52057 Aachen, Germany
e-mail: (hklu{at}pcserver.mk1.rwth-aachen.de)


   Abstract
Top
 Abstract
Introduction
 Comment
 References
 
Anomalous origin of the right coronary artery from the main pulmonary artery is a rare congenital cardiac malformation. Most patients remain asymptomatic. However, there are cases of sudden cardiac death described in the literature, indicating a potentially malign course of the disease. To establish a double-ostium coronary system, correction of the aberrant vessel is recommended. Despite surgical reconstitution of normal coronary anatomy, the postoperative clinical presentation of some patients does not improve substantially, raising the question of the functional outcome of reinserted coronary vessels. This report of a patient with anomalous origin of the right coronary artery from the pulmonary trunk, in whom a complete hemodynamic assessment including intracoronary Doppler flow measurements was performed before and after reimplantation, very strongly supports the concept of an anatomically corrective operation.


   Introduction
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 Abstract
 Introduction
Comment
 References
 
A 60-year-old woman (height 164 cm, weight 68 kg) was first angiographically diagnosed to have an anomalous origin of the right coronary artery (RCA) from the pulmonary trunk (PA) when she was referred to the hospital in 1988 with dyspnea, New York Heart Association (NYHA) functional class II. She was followed up on a regular basis until autumn 1996 when she reported progressive dyspnea during exercise (now NYHA functional class III) chronic fatigue, and angina with exertion. Physical examination revealed a blood pressure of 120/65 mm Hg and a pulse rate of 55 beats per minute. The first and second heart sounds were of normal intensity without audible heart murmurs. The remaining physical examination was normal. Electrocardiogram showed sinus rhythm with incomplete left bundle-branch block. Chest roentgenogram revealed a cardiacthoracic ratio of 0.4 and a slightly prominent left pulmonary segment. Two-dimensional echocardiogram demonstrated a dilated RCA that appeared to originate from the posterior wall of the main pulmonary artery and course toward the septum. Doppler examination revealed a localized turbulent diastolic flow directed into the main pulmonary artery adjacent to the origin of the RCA. During treadmill exercise testing, the patient developed complete left bundle-branch block at 50 W. Thallium 201 exercise test documented homogeneous left ventricular perfusion.

Combined right and left heart catheterization including intravascular ultrasound and Doppler was performed to assess the hemodynamical significance of the coronary malformation. Global ejection fraction was 0.69 without regional wall motion abnormalities. Selective coronary angiography confirmed the previously described malformation with a large dilated and tortuous left coronary artery originating from the left coronary sinus and typical course of left anterior descending coronary artery (LAD) and left circumflex artery (LCX) (Fig 1A). The RCA was contrasted through several large epicardial collaterals originating from the LAD and LCX. The vessel itself was also dilated and drained into the pulmonary trunk. Transfer of contrast dye through the collaterals was extremely fast and occurred almost simultaneously with the periphery of the LCX (Fig 1B). It was not possible to fill the coronary sinus with contrast medium during the late venous phase of the coronary angiogram. Blood samples were obtained from various positions including the middle third of the RCA (through a support catheter). All pressure, Doppler flow, intravascular ultrasound, and blood saturation measurements are given in Table 1. Systemic cardiac output was calculated to be 5.7 L/min with a pulmonary to systemic flow ratio of 1.14:1, leaving a left to right shunt of 0.8 L/min through the left coronary artery. The contribution of the coronary blood flow to the systemic cardiac output was increased to 18% at rest (normal, 3% to 5%).



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  		Fig 1. Selective coronary angiography of a patient with anomalous right coronary artery originating from the pulmonary artery before (A, B) and after (C, D) reinsertion of the aberrant vessel into the aorta. (A) Contrast injection into the left main coronary artery, early phase of the angiogram. The left coronary artery is dilated and tortuous, with typical course of the left anterior descending artery (LAD) and the left circumflex artery (LCX). (B) Contrast injection into the left main coronary artery, late phase of the angiogram. The right coronary artery (RCA) receives contrast material through several large epicardial collaterals mainly originating from the LCX. The RCA itself is dilated and drains into the main pulmonary artery (MPA). One additional catheter is placed in the ostium of the RCA. (C) Postoperative angiogram with contrast injection into the left main coronary artery. The LAD and LCX have substantially reduced their luminal diameters (about 50%) without visualization of shunting collaterals. (D) Postoperative angiogram with contrast injection into the RCA originating from the ascending aorta (AoA). Antegrade flow and normal runoff into the periphery, obviously draining into a well-developed capillary bed, are documented. The luminal diameter of the RCA has been reduced by 17%.

 

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  		Table 1. Preoperative and Postoperative Pressure and Blood Saturation, Cardiac Output, Doppler Flow, and Quantitative Coronary Angiography Measurements in a Patient With Anomalous Origin of the Right Coronary Artery From the Pulmonary Trunk

 
Both coronary flow velocities and flow patterns obtained with a 0.014-inch Doppler guidewire in the LAD were abnormal, showing a balanced diastolic to systolic flow velocity ratio of 0.9 with very high baseline velocities (average peak velocity: 59 cm/s) (Fig 2). Intravascular ultrasound confirmed the large vessel diameters and areas without signs of coronary artery disease. The RCA demonstrated a veinlike collapsing phenomenon during early systole (maximum area: 45 mm2, minimum area: 25 mm2, measurements at the very proximal portion of the vessel). Coronary flow reserve after intracoronary injection of 12 mg of papaverine hydrochloride documented a ratio of 1.2 in the LAD.



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  		Fig 2. Doppler flow measurements obtained with a 0.014-inch Doppler wire of the left anterior descending artery (LAD) in a patient with anomalous right coronary artery originating from the pulmonary artery before (A) and after (B, C) reinsertion of the aberrant vessel into the aorta. (A) Documentation of a balanced diastolic to systolic flow velocity ratio of 0.9 and very high baseline flow velocities [average peak flow velocity (APV = 59 cm/s)] in combination with a low coronary flow reserve ratio of 1.2. (B) Postoperative documentation of a diastolic to systolic flow velocity ratio of 1.7 in combination with normalized baseline flow velocities (APV: 16 cm/s). (C) Postoperative peak flow velocity in the LAD (APV: 53 cm/s) after intracoronary application of 12 mg of papaverine hydrochloride with a normalized coronary flow reserve ratio of 3.8.

 
Surgical reimplantation of the RCA into the aorta was performed under moderate hypothermia and cardioplegic arrest while the patient was maintained on cardiopulmonary bypass. Before the RCA was excised, the right atrium was explored and a well-developed coronary sinus was seen. The anomalous right coronary artery was excised with a button of the pulmonary artery and reimplanted anteriorly into the aortic root. The hole in the pulmonary artery was closed with a pericardial patch. The postoperative course was uncomplicated.

Fourteen weeks after the operation the patient was readmitted for clinical reevaluation and control angiography. She reported persistent dyspnea, now NYHA functional class I to II, without fatigue. Right heart catheterization documented normal pulmonary pressures without oxygen step-up. Coronary angiography revealed antegrade flow in the RCA with a normal runoff into the periphery, obviously draining into a well-developed capillary bed. LAD and LCX showed antegrade flow without visualization of shunting collaterals. The coronary sinus was now normally visualized during the venous phase of the coronary angiogram. LAD and LCX had substantially reduced their diameters (about 50%) without being tortuous (Fig 1C). The mean lumen diameter of the RCA had been reduced by 17% (Fig 1D). Coronary flow reserve after papaverine hydrochloride provocation was normal in all vessels (LAD: 3.3; LCX and RCA: 3.8).


   Comment
Top
 Abstract
 Introduction
 Comment
References
 
This report describes a complete preoperative and postoperative invasive hemodynamic evaluation of the coronary pathophysiology in a patient with anomalous origin of the RCA from the pulmonary artery and reimplantation of the aberrant vessel into the aorta. Preoperative flow through the left coronary artery was increased to 18% of the cardiac output with a left-to-right shunt of 0.8 L/min and a pulmonary to systemic flow ratio of 1:1.14. The coronary flow reserve determined by intracoronary papaverine hydrochloride application was severely reduced (ratio; 1.2) and the most likely cause for clinical symptoms with exertion. The described coronary pathophysiology of a high flow velocity (59 cm/s) in combination with a low coronary flow reserve was most likely due to a flow-induced dilatation of the coronary vessels including the resistance vessels at rest. Therefore, increasing the oxygen demand led to functional exhaustion of the physiologic vascular compensatory mechanisms.

After the operation, the clinical presentation of the patient improved slowly but substantially. Control heart catheterization documented antegrade flow in the RCA and an about 40% reduction in size of all coronary vessels. More impressive, coronary flow reserve measurements revealed a normal vasomotor response to pharmacologic stimulation indicating complete functional recovery of the preoperatively dilated coronary arteries. Reestablishment of the coronary anatomy was accompanied by normal coronary flow and complete functional recovery.

Origin of the right coronary artery from the pulmonary artery is a rare congenital malformation with a potentially malignant character. Of 57 previously reported cases (Table 2), the abnormal vessel was first diagnosed during angiography in 30 patients [12–14]. The age of the patients when the abnormality was recorded ranged from 1 day to 90 years [5, 38]. As many patients remain asymptomatic (41% of the patients in this database) [12, 13, 28, 35] the true incidence of this coronary malformation might be higher. This is clearly in contrast to the Bland–White–Garland syndrome with anomalous origin of the left coronary artery from the pulmonary artery, in which patients early in childhood often show mitral insufficiency and signs of anterolateral myocardial infarction as early manifestations [50]. When symptomatic, the clinical presentation of patients with an anomalous RCA is nonuniform, including dyspnea (17%) [18, 23, 41], fatigue (13%) [18, 19], congestive heart failure (30%) [14, 22, 32, 42], angina (17%) [26, 36, 49], myocardial infarction (9%) [31, 32], and even cardiac arrest (17%) [17, 38]. The objective findings are, except for the morphologic coronary angiography, nonspecific in most cases; objective signs of ischemia developed in 40% of the patients [15, 26, 31, 49]. The only possible sign of ischemia in our patient was conversion from incomplete to complete left bundle-branch block during exercise testing.


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  		Table 2. Review of Literature Including Total of 57 Patients With Anomalous Origin of Right Coronary Artery From Pulmonary Trunk

 
Associated congenital cardiac defects are reported in 40% of the patients with abnormal origin of the RCA [1, 6, 40, 48], an aortopulmonary window, being the most commonly associated malformation (30% of all patients with associated defects) [15, 22, 37]. Tetralogy of Fallot (19%) [9, 10] and isolated valvular defects [27, 32] have been described as well.

In general, a corrective operation is recommended in this cardiac malformation even for asymptomatic patients, and was performed in 87% of those patients diagnosed during their lifetime (75% in this database), mainly for two reasons. First, with reimplantation of the aberrant vessel into the aorta, a double ostium coronary system is reestablished with a potentially lower risk for sudden cardiac death. Second, the operation provides relief from coronary (pulmonary) steal, which may be responsible for myocardial ischemia [26]. Simple ligation of the aberrant vessel at its origin to abolish the pulmonary steal should be recognized as a less traumatic surgical alternative with the persistent risk of a single ostium coronary system [23]. Only a few patients have been treated medically, with no clinical follow-up available [29, 36].

Clinical or angiographic follow-up after a corrective operation was available for 9 patients [16–20, 28, 45, 49]. A reduction in the size of the LAD after reimplantation of the RCA into the aorta was observed before [28]. Persistent symptoms and myocardial ischemia were described in two patients with postoperatively dilated coronary arteries and slow runoff into the periphery after reimplantation [49]. In our patient both complete morphologic and functional recovery of the coronary vasculature with clear clinical improvement was achieved by an anatomical corrective operation.

Unlike in Bland–White–Garland syndrome, in which translocation of the left coronary artery is mandatory [51], medical treatment should be recognized as an alternative therapeutical approach for anomalous origin of the right coronary artery from the pulmonary artery. Yet this systematic evaluation of the coronary pathophysiology in anomalous origin of the RCA from the pulmonary artery before and after reinsertion of the aberrant vessel into the aorta very strongly supports the therapeutical concept of an anatomic corrective operation.


   References
Top
 Abstract
 Introduction
 Comment
 References
 

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