C syndromes, including sudden death. Krause [ 11

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1 CURRENT REVIEW Surgical Therapy in the Management of Coronary Anomalies: Emphasis on Utility of Internal Mammary Artery Grafts Amram J. Cohen, MD, MAJ, Brent A. Grishkin, MD, COL, Robert A. Helsel, MD, COL, and Harold D. Head, MD, COL (RET), All MC, USA Department of Surgery, Cardiothoracic Surgery Service, Brooke Army Medical Center, Fort Sam Houston, Texas, and Valley Children s Hospital, Fresno, California Three patients with an anomalous main coronary artery coursing between the great vessels are presented with a review of the literature. Their surgical treatment by coronary artery bypass grafting with use of the ipsilateral internal mammary artery is described with angiographic follow-up. Young patients with these anomalies, whether they are asymptomatic or have syncope, are at high risk for sudden death. Older patients with angina appear to be adequately risk stratified by thallium stress tests. For patients requiring surgical intervention, aortoplasty and coronary artery bypass grafting have both resulted in relief of symptoms, but the follow-up is limited. We suggest that coronary artery bypass grafting with an ipsilateral internal mammary artery graft to the anomalous vessel is the procedure most likely to relieve ischemia and achieve good long-term results for both symptomatic relief and survival. ( 1989;47:630-7) oronary anomalies can be associated with clinical C syndromes, including sudden death. Krause [ 11 described a coronary arteriovenous fistula in By 1970, Ogden [2] had collected 224 cases of coronary anomalies and classified them into major, minor, and secondary types. According to Ogden, major coronary anomalies have either an abnormal communication between a coronary artery and a cardiac chamber or an abnormal origin of the coronary artery from the pulmonary artery. In a secondary anomaly, the coronary artery variation represents a circulatory response to primary intracardiac pathology. In a minor coronary anomaly, there is variation of the origin of the vessels from the aorta, but the distal circulation is normal. Minor anomalies represent one third of all coronary anomalies [Z]. Most of them are benign variants of normal anatomy. However, two minor anomalies-the left main coronary artery (LMCA) arising from the right aortic sinus and the right main coronary artery (RCA) arising from the left aortic sinus (LAS)-have been associated with both clinical symptoms and death [3]. In normal anatomy, the LMCA has its ostium in the LAS and courses between the pulmonary artery anteriorly and the left atrium posteriorly for an average distance of 10 to 20 mm before reaching the left atrioventricular groove and bifurcating into the left anterior descending and circumflex coronary arteries. The RCA originates from the right aortic sinus and courses directly into the right atrioventricular groove. Address reprint requests to Dr Cohen, Cardiothoracic Surgery Service, Walter Reed Army Medical Center, Washington, DC The opinions or assertions contained herein are the private views of the authors and are not to be construed as reflecting the views of the Department of the Army or the Department of Defense. In anomalous origin of the RCA from the LAS, the RCA will course anteriorly and to the right, and will almost always run between the pulmonary artery anteriorly and the aorta posteriorly (Fig 1A). With anomalous origin of the LMCA from the right aortic sinus, the LMCA can take one of four different paths to reach the left atrioventricular groove. It can go anterior to the pulmonary artery, posterior to the aorta, or between the great vessels, or it can take a septa1 course through the conal septum (Fig 1B) [4]. The latter two courses and the anomalous RCA have been the minor anomalies most often associated with serious clinical symptoms. This report describes patients treated surgically for these anomalies. Material and Methods The files of the Thoracic Surgery Service at Brooke Army Medical Center from July 1984 through December 1986 were reviewed to locate patients with abnormal origin of a main coronary artery that coursed between the great vessels. Chart review documented presenting symptoms and signs, diagnostic procedures used, and operative findings. Follow-up for all patients was obtained by postoperative stress testing, angiography, and an interview by one of us. Results There were 3 patients in our series, all men, aged 38, 48, and 51 years. Two patients were seen with angina pectoris; 1 had exertional angina, and the other had angina both with exertion and at rest. The 38-year-old patient was seen with exertional syncope. Two patients had no prior cardiac history; the third had a history of paroxysmal atrial fibrillation.

2 1989;47:63&7 COHEN ET AL 631 CF A Fig 1. Anomalies of the main cororiar!/ arteries: (A) origin of right coronar!/ artery (RCA) from the left aortic sinus and (B) possible courses of the left main coronary urter!/ (LM) arising from the right coronary sinus; LM, = left main coronary artery coursing posterior to the aorta; LM, = left main coronar!/ arterq coursing betrueen thc aorta (Ao) and prrlmonary artery (Pa); LM, = left main coronary artery with srptal course through the outflor~~ tract of the right ventricle; arid LM, = left main coronny artery coursing anterior to the pulmonary artery. (CF = circumflex coronar!/ artery; LAD = left anterior descending coronary artmy.) B All 3 patients underwent an exercise thallium stress test and angiography preoperatively. Two patients had an anomalous LMCA arising from the right aortic sinus. One of them had associated coronary artery disease (CAD) (Fig 2); the exercise stress test indicated ischemia, and the thallium stress test showed reversible ischemia (Fig 3). The other patient had no associated CAD (Fig 4). His exercise stress test showed no ischemia at maximum exercise, but a thallium stress test demonstrated reversible ischemia. The third patient had an anomalous RCA arising from the LAS with no associated CAD (Fig 5). The results of his exercise stress test were normal, but the thallium stress test showed reversible ischemia. Both patients with an anomalous LMCA were treated with ipsilateral internal mammary artery (IMA) grafts to the left anterior descending coronary artery and reverse saphenous vein grafts (RSVGs) to the circumflex system. In 1 of these patients in whom the origin of the anomalous vessel was dissected, the LMCA originated perpendicular to the aorta. The patient with an atherosclerotic lesion of Fig 2. Left anterior oblique view of the left coronary artery system. The angiography catheter (C) is in the right aortic sinus. The left main coronary artery (LM) arises from the right aortic sinus. The arrow demonstrates the atherosclerotic lesion in the diagonal coronary artery. Fig 3. Thallium stress test in a patient with anomalous left main coronary artery. Circumferential quantitative analysis of the third, fourth, and fifth slices shows only the inferoseptal uull (from 180 to 270 degrees) has washout values in the nori7lul range (above the light blue band on each graph). The entire left coronary artery system displays reversible ischemia. Stress, redistribution, and washout curues are color-coded white, yellow, and pink, respect iuely.

3 632 COHEN ET AL 1989; Fig 4. Right anterior oblique view dertiotistratirig origiri of Hie left main coronary artery (LM) arid the right cororiar!/ artery (RCA) frorii a cotiinion origiti (arrow) in the right aortic siriris. a diagonal branch had this artery bypassed with an RSVG. In the patient with the anomalous RCA, the RCA originated from the LAS adjacent and anterior to the LMCA. The RCA was intimately associated with the aortic wall for 1.5 cm in its course between the aorta and the pulmonary artery, and then continued along the right atrioventricular groove. In this patient, the right IMA was used to bypass the RCA in its proximal portion with ligation of the origin of the RCA (Fig 6). One patient required reexploration for mediastinal hemorrhage. Follow-up ranged from 9 to 14 months. At Fig 5. Left anterior oblique view dernonstrating fhe angiography catheter (C) in the left aortic sinus. The right coronary artery (RCA) is seen to arise from the left aortic sinus. Fig 6. Left aiiterior oblique view of the anastornosis of the right interrial niairirriary artery (IMA) with the right coronary artery (RCA). The arrow rtmrks the area of anastortiosis. follow-up, all 3 patients were asymptomatic, had normal thallium stress tests, and had angiographically patent IMA grafts. The RSVG to a diagonal branch was occluded at follow-up. Comment In 1962, Jokl and colleagues [5] described the lethal potential of minor coronary anomalies with the autopsy finding of an anomalous origin of the LMCA in a young athlete who died suddenly. In 1974, Cheitlin and coworkers [6] described a young man with an RCA anomaly and otherwise normal coronary arteries who had an inferior myocardial infarction (MI). They also described a 14-year-old boy who sustained an MI related to exertion and who had an anomalous LMCA treated surgically with an aortoplasty; 9-year follow-up showed no further exertional symptoms [7]. In 1983, Bloomfield and associates [8] reported the surgical treatment of an anomalous RCA; a 58-year-old man with intractable angina and otherwise normal coronary arteries obtained relief of symptoms after coronary artery bypass grafting (CABG). A review of the literature supports our aggressive therapeutic approach in symptomatic older patients and all younger patients with these anomalies. Eighty-seven autopsy cases in which an anomalous main coronary artery passed between the great vessels have been reported. Forty-six involved an anomalous LMCA; among these, 36 of the deaths were clearly coronary in origin [ Twenty-nine of these 36 patients were less than 30 years of age. Twenty-eight of them died suddenly, 1 had

4 1989; COHEN ET AL 633 Table 1. Preoperative Noninvasive Testing for Patients With Anomalous Main Coronary Artery and Angina" Anomalous Graded Exercise Thallium Coronary Artery Reference Year Age (yr) Sex Vessel Symptom Stress Test Stress Test Disease Chaitman et a1 [29] M LMCA Angina Positive Not done Present Thompson et a1 [32] M RCA Angina Positive Not done Absent 56 M RCA Angina Nondiagnostic Not done Absent Moodie et a1 [25] M LMCA Angina Positive Not done Present Keren et a1 [36] M RCA Angina Positive Not done Present Bloomfield et a1 [8] M RCA Angina Postive Positive Absent Liberthson et a1 [17] F LMCA Angina Nondiagnostic Positive Present Donaldson et a1 [21] Mb LMCA Angina Positive Not done Absent... Mb LMCA Angina Positive Not done Absent Grey et a1 [22] F LMCA Angina Not done Positive Absent Iskandrian et a1 [23] M LMCA Angina Positive Positive Absent a In 1979, Liberthson and associates (241 described 6 patients who were seen because of angina and an LMCA anomaly. Four exercise stress tests were positive, and two thallium stress tests were positive. Four of these patients had associated coronary artery disease. No other data are available. Ages of the patients in this series were 15, 21, and 32 years, and could not be assigned to individual patients. LMCA = left main coronary artery; RCA = right coronary artery. an MI before death, and none had associated CAD [9, 13, 141. Of the 7 patients older than 30 years, 5 had associated CAD, and 3 had a documented MI before death [6,15]. Of the evaluated patients, half of the young patients had symptoms before death, and all patients older than 30 years had symptoms before death. In the younger patients, the symptoms were evenly distributed between syncope and angina, whereas in the older group the symptoms were predominantly angina or MI [3, 6, 9, 13-15]. Of the 41 patients with an anomalous RCA, 8 died of heart-related causes, and all 8 died suddenly [13, 16-19]. Seven of these 8 patients were younger than 30 years, and none had associated CAD or MI before death. Two young patients had prior symptoms, syncope in 1 and angina in the other [16, 191. For both anomalies, younger patients died suddenly, without associated CAD, and half had been asymptomatic before the fatal event. Symptoms, when present, were evenly distributed between syncope and angina. Older patients had associated CAD and anginal symptoms, and half had a documented MI. Eighty-three patients with a main coronary anomaly coursing between the great vessels have been diagnosed antemortem. Of this group, 37 had LMCA anomalies, and the patient profile was similar to those who died suddenly [4, 7, Young patients were seen mostly with syncope and without associated CAD. The older patients Table 2. Preoperative Noninvasive Testing for Patients With Anomalous Main Coronary Artery and Syncope Reference Year Age Anomalous Graded Exercise Thallium Coronary Artery (yr) Sex Vessel Symptoms Stress Test Stress Test Disease Cheitlin et a1 [6] M LMCA Syncope Normal Not done Absent Pedal [37] F LMCA Syncope Normal Not done Absent Murphy et a1 [28] F LMCA Syncope Normal Not done Absent Benge et a1 [33] M RCA Syncope Normal Nondiagnostic Absent Mustafa et a1 [26] M LMCA Syncope Normal Normal Absent Donaldson et a1 [21] a M LMCA Syncope Positive (ventricular Not done Absent tachycardia) Brandt et a1 [34] M RCA Syncope Normal Not done Absent (ventricular tachycardia) Bett et a1 [35] M RCA Syncope Positive (ventricular Negative Absent tachycardia) Barth and Roberts [9] M LMCA Syncope/angina Normal Not done Absent a Ages of the patients in this series were 15, 21, and 32 years, and could not be assigned to individual patients LMCA = left main coronary artery; RCA = right coronary artery.

5 634 COHEN ET AL 1989;4763&7 usually had associated CAD, with angina as a predominant symptom. Although the majority of the patients diagnosed at autopsy were less than 30 years old, the majority of the patients seen clinically were more than 30 years old, a finding suggesting that patients seen at an early age frequently have sudden death as the initial presentation. Among the 46 patients with an anomalous RCA, 19 were of unknown age. Of the remaining 27, only young patients were seen with syncope; the older ones predominantly had angina. A preponderance of young patients in the autopsy series and of older patients in the clinical series was again present [6, 8, 24, 29-36]. Stress testing to assess ischemic consequences of these anomalies has had limited success. Results of such tests have been reported for 20 patients with an anomalous main coronary artery coursing between the great vessels (13, LMCA; 7, RCA). Patients who had, with or without associated CAD on angiography, had positive exercise stress tests indicative of ischemia. In the few patients in whom these stress tests were equivocal or not reported, the thallium stress test was positive (Table 1). In contrast, in patients seen with syncope or arrhythmias, neither stress test demonstrated ischemia. The positive stress tests in this group were found only in patients in whom ventricular tachycardia developed at maximum exercise (Table 2). Two of our patients were seen with angina, with ischemia detected by stress test. To our knowledge, our patient seen with syncope represents the first such patient in whom a thallium stress test demonstrated reversible ischemia. For patients with these anomalies, the operative procedure chosen depends on the mechanism of ischemia. In theory, this anomaly can be associated with an acute origin of the anomalous vessel causing a slitlike orifice and ostial obstruction during exercise [8, 361. Alternatively, ischemia can be caused by compression of the anomalous artery between the great vessels during exercise [7, 381. In the first instance, a procedure to enlarge the ostium should relieve ischemia. Such an aortoplasty has been performed in 8 patients with anomalous LMCAs (Table 3). All were symptomatic, and all were relieved of symptoms. Follow-up ranged to 9 years, and five vessels were proved patent by postoperative angiography. In 6 patients, the anomalous ostium was relocated so that it originated from the LAS [21, 261. In the other 2 patients, the orifice was simply enlarged to compensate for the acute angle of takeoff [6, 221. Although this operation has yielded good results, it has limitations. First, in some patients, there is no acute takeoff of the anomalous vessel, and an ostioplasty would not be an appropriate procedure. Second, if a component Table 3. Anomalous Left Main Coronary Artery Treated Surgically by Aortoplasty Reference Cheitlin et a1 [6] Mustafa et a1 [26] Donaldson et a1 [21] Grey et a1 [22] FoIIow-u~ Age Coronary Artery Year (yr) Sex Symptoms Procedure Disease Length Result M M F M Ma M M F syncope, shortness of breath, exertional syncope Nonexertional Aortoplasty Absent 9 Y Aortoplasty Absent 2 mo Ascending aortic replacement, aortic valve replacement Aortoplasty Absent Absent 3 Y 2 Y No data Asymptomatic aortic insufficiency, angiographicall y patent left main coronary artery Angiographically patent left main coronary artery Asymptomatic clinically Stress test normal No data a Aortoplasty Absent Aortoplasty Absent a Aortoplasty Absent Aortoplasty Absent No data No data a Patients in this series were 15, 21, and 32 years of age. They could not be identified individually. Follow-up ranged from 10 to 24 months. There were no clinical symptoms, and postoperative angiography showed a patent left main coronary artery in all 3 patients.

6 1989;47 63G7 COHEN ET AL 635 Table 4. Reported Surgical lnteruention for Anotnalous Left Main Coronary Artery" Reference FOIIOW-UP Age Coronary Artery Year (yr) Sex Symptoms Grafts Disease Length Result Sachs et al [27] M Ventricular RSVGlleft Absent 5 mo fibrillation main Murphy et a F Syncope RSVGILAD Absent 1 Y' Liberthson et a1 [17Ih F Chest RSVGILAD, pain at circumflex Absent 1 Y' rest Ishikawa and Brandt [4] M Angina No data Present No data 73 M Angina No data Present No data Iskandrian et a1 [23] M Angina RSVGILAD, Present 8 wk circumflex, right coronary artery Angiographically patent vein graft; stress test normal Angiographically patent vein graft Angiographically patent vein graft No data No data Normal thallium stress test This table includes saphenous vein bypass grafting only. In 1979, Libcrthson and colleagues [24] described 2 patients who underwent coronary artery bypass grafting for refractory angina with this anomaly without follow-up. LAD = left anterior descending coronary artery; RSVG = reverse saphenous vein graft. of ischemia is caused by compression of the anomalous artery between the great vessels, an ostioplasty alone will not completely relieve ischemia. Third, detachment of the intercoronary commissure necessary during this procedure can cause aortic insufficiency [7]. Finally, there is potential for rapid progression of atherosclerosis at the ostium of the instrumented vessel. An alternative procedure for this anomaly is CABG with either RSVGs or IMA grafts. This procedure has been performed with RSVGs in 11 patients, 6 with an anomalous LMCA and 5 with an anomalous RCA. The 6 patients with an anomalous LMCA all had relief of symptoms after the operation with follow-up to 1 year (Table 4). Two patients were seen with abnormal stress tests that reverted to normal, and repeat catheterization in 3 patients showed patent vein grafts. Five patients underwent operation for an anomalous RCA using an RSVG to the RCA at the base of the right atrium (Table 5). All patients had relief of symptoms with follow-up to 39 months. Stress tests in 2 patients reverted to normal. These limited results with RSVG CABG for an LMCA originating from the right aortic sinus or an RCA originating from the LAS appear good. However, long-term results may be less favorable. Reverse saphenous vein Table 5. Reported Surgical Intervention for Anomalous Right Coronary Artery Age FOIIOW-UP Reference Year (yr) Sex Symptoms Grafts Cardiac Disease Length Result Kirnbiris et a1 [30] M Ventricular No data fibrilla tion Keren et al M Angina No data Bloomfield et a1 [8] M Angina Reverse saphenous vein graftiright coronary artery Brandt et a1 [34] M Myocardial Reverse cephalic infarction, vein graftiright ventricular coronary artery fibrillation Bett et a M Syncope Reverse saphenous vein graftkight coronary artery Absent No data No data Present No data No data Absent 2 mo Stress test normal, no ischemia Absent 9 mo Stress test normal, no symptoms Absent 39 mo Running 128 kmlwk without symptoms

7 636 COHEN ET AL 1989;4763&7 grafts to minimally diseased vessels show a patency rate similar to that of grafts placed to stenosed arteries, but with acceleration of atherosclerosis in the native vessel [39, 401. Reverse saphenous vein grafts to nonatherosclerotic vessels have achieved poor results. Late follow-up of such grafts placed to correct major coronary anomalies shows one third of the grafts occluded, with the majority having either aneurysmal dilation or stenosis at 6 months [41]. Late closure of grafts to major vessels in young patients without collateral channels can have severe consequences. The IMA was used previously by Moodie and colleagues [25] for an anomalous LMCA, and our 2 patients constitute the fourth and fifth such applications of IMA CABG. Our use of the right IMA to bypass the RCA is the first such reported instance of which we are aware. All the patients in the series of Moodie and associates were symptomatic preoperatively, and all were relieved of symptoms by the procedure. These results combined with our own suggest a good short-term outcome for this procedure. Long-term patency of IMA grafts exceeds that for RSVGs. In minimally diseased vessels, the patency rate for the IMA graft is better than that for the RSVG, and there is less chance of progression of proximal disease in the native vessel [39]. Patency rates achieved by Laborde and associates [42] with native subclavian artery grafts to treat major coronary anomalies suggest that arterial grafts will remain open when grafted to nondiseased arteries. We propose that a patient found to have an anomalous main coronary artery coursing between the great vessels be treated in the following manner. Patients less than 30 years of age should undergo an operation because of the high incidence of sudden death as the initial presentation in younger patients. For patients more than 30 years of age, the presenting symptoms are relevant. Patients with syncope should have an operation regardless of the results of stress testing. Patients with should undergo angiography to evaluate indications for a cardiac operation, and if such indications are present, the operation should include CABG of the anomalous system. If the angiogram shows no other vessels needing bypass grafting, the anomaly should be bypassed only if thallium stress testing shows reversible ischemia. If the thallium stress test is negative, the patient can be observed, as sudden cardiac death is less likely in older patients. If the patient is asymptomatic, he or she can either be observed or undergo thallium stress testing with therapy dependent on the presence or absence of reversible ischemia. We believe the procedure of choice for this anomaly, when operation is necessary, is CABG with the ipsilateral IMA grafted to the anomalous coronary system. Questions regarding CABG with the IMA as a procedure for this anomaly remain unanswered. (1) Should patients with an anomalous LMCA undergo a separate graft to the circumflex system? (2) Should the anomalous vessel be ligated proximally? In patients with an anomalous LMCA, we recommend bypass grafting of the circumflex system because compression of the left coronary system can occur distal to the bifurcation of the LMCA. We suggest an RSVG be used in preference to the right IMA graft to bypass the circumflex coronary artery be- cause of the difficulty encountered at repeat sternotomy when a right IMA graft has been placed to the left coronary system, and because of the unknown patency rate of the right IMA to the circumflex system. In young patients, in whom long-term results are unknown, a repeat sternotomy must be considered a possible future event. 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