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a Division of Cardiovascular Surgery, Mayo Clinic, Rochester, Minnesota
b Division of Cardiology and Cardiovascular Diseases, Mayo Clinic, Rochester, Minnesota
Accepted for publication June 4, 2009.
* Address correspondence to Dr Burkhart, Division of Cardiovascular Surgery, Mayo Clinic College of Medicine, 200 First St SW, Rochester, MN 55905 (Email: burkhart.harold{at}mayo.edu).
Presented at the Forty-fifth Annual Meeting of The Society of Thoracic Surgeons, San Francisco, CA, Jan 26–28, 2009.
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Abstract |
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Methods: A retrospective review of 36 patients (23 male) who underwent operation between October 1992 and August 2008 for AAOCA was performed. Median age was 47 years (range, 13 to 82 years). Angina, shortness of breath, or syncope was present in 29 (81%), and 9 of 21 (43%) had an abnormal stress test. Coronary or computed tomographic angiography demonstrated an anomalous left main coronary artery arising from the right sinus in 13 (36%), right coronary artery arising from the left sinus in 21 (58%), and left anterior descending artery arising from the right sinus traveling between the aorta and pulmonary artery in 2 (5%). An intramural course was identified on preoperative imaging in 34 (94%). Although no patients had significant associated atherosclerotic coronary artery disease, 5 (14%) had previous acute myocardial infarction related to the AAOCA.
Results: Operation included coronary artery bypass grafting in 14 patients and unroofing in 22; 6 patients had associated cardiac procedures performed. There were no early deaths. There was one late death secondary to a subdural bleed. At follow-up (mean 1.1 years; maximum 14 years), chest pain recurred in 1 patient who had coronary artery bypass grafting. No recurrent symptoms were noted in the unroofing group.
Conclusions: Unroofing of an anomalous coronary artery can be performed safely with excellent results in the majority of patients. When concomitant atherosclerotic coronary artery disease is present, coronary artery bypass grafting is an appropriate alternative.
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Introduction |
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Several surgical techniques have been described to treat AAOCA including coronary artery bypass grafting (CABG), unroofing of the anomalous coronary artery, reimplantation of the anomalous coronary artery, and pulmonary artery translocation [9–16]. Our practice at the Mayo Clinic has evolved from CABG to unroofing of the anomalous coronary artery. In the case of associated coronary artery disease, CABG ± unroofing has been employed. The following is a retrospective review of our experience with AAOCA and current management strategy.
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Material and Methods |
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A total of 50 patients were identified who underwent surgical repair of AAOCA. Fourteen of these patients were eliminated because they had multivessel atherosclerotic coronary artery disease as the indication for surgery and were treated only with coronary CABG. Therefore, the study group consisted of the remaining 36 patients treated primarily for AAOCA. The median age was 47 years (range, 13 to 82 years) and 23 (64%) patients were males. Preoperative characteristics are shown in Table 1. Symptoms were present in 29 (81%) patients and included chest pain in 20 patients, shortness of breath in 7, and syncope in 2. Twenty-one patients had preoperative stress tests, of which only 9 were abnormal. Preoperative imaging (coronary angiography or computed tomographic angiography) demonstrated the left main coronary artery arising from the right coronary sinus (ALCA) in 13 (36%) patients, the right coronary artery arising from the left coronary sinus (ARCA) in 21 (58%), and the left anterior descending artery arising from the right coronary sinus (ALAD) in 2 (5%). There was no associated significant coronary artery disease in any of the patients. Five patients had a history of acute myocardial infarction secondary to the anomalous coronary (2 ALCA, 2 ARCA, and 1 ALAD). In all the patients, the anomalous coronary artery arose from the opposite sinus and traveled between the aorta and pulmonary artery. An intramural course was identified in 34 (94%) of patients and was unknown in 2 (6%) of patients. One patient had previously undergone surgical repair of an ARCA with a right internal mammary artery to the right coronary artery bypass using a daVinci robotic technique (Intuitive Surgical, Sunnyvale, CA) at another institution. This patient presented with recurrent chest pain and an atretic internal mammary artery.
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Excess tissue was removed and the remaining edges were tacked down with fine suture. If the intramural pathway passed under the superior aspect of the right-left aortic valve commissure, the commissure was taken down and then resuspended. If the intramural pathway was lower and there was concern for potential aortic valve damage, the AAOCA was unroofed only in its true sinus, creating a neo-ostium while leaving the pathway deep to the valve intact.
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2 tests as appropriate. Vital status for all patients was attained through use of Accurint (http://www.accurint.com) and the Social Security Death Index. Kaplan-Meier survival curves were created using SAS version 9.1 (SAS, Cary, NC) and compared using a log-rank test. |
Results |
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Mean follow-up was 1.1 ± 2.8 years and maximum follow-up was 14 years. At last follow-up, vague chest pain was present in only 1 patient originally treated with CABG (1999) using bilateral internal mammary arteries (IMAs) for ALCA. Coronary angiogram performed 6 months postoperatively revealed patent small IMAs with good left native coronary flow. The one patient with recurrent chest pain previously treated with a right IMA for ARCA at another institution had an unroofing performed at our institution with resolution of symptoms. No patients treated with unroofing of either anomalous coronary artery had recurrent symptoms. Late interventions included catheter ablation for atrial fibrillation in one patient and aortic valve homograft placement for aortic valve endocarditis in one. One patient who underwent a redo aortic valve replacement and unroofing of an anomalous right coronary artery suffered a postoperative subdural hematoma and subsequently died at a rehabilitation center two months later.
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Comment |
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Many patients are asymptomatic at the time of presentation or diagnosis [3–8]. Unfortunately, the first symptom may be death. Several studies have shown clearly that ALCA is associated with a higher mortality [4–8]. Sudden death has also been described in patients with ARCA [2, 3, 6–8]. Taylor and colleagues [3] and Basso and colleagues [8] both showed that sudden death was more common in ALCA than ARCA, but each anomaly is associated with sudden death.
Although CABG has been used by many for surgical treatment of AAOCA, concerns regarding the internal mammary artery exist [17, 18]. Given that many patients with AAOCA may have adequate coronary blood flow most of the time, competitive flow may keep the IMA from maturing. Fedoruk and colleagues [17] reported on 5 patients treated with right IMA bypass graft for ARCA. Two of these 5 patients ended up with graft occlusion. Tavaf-Motamen and colleagues [18] described 4 patients with surgical corrected ARCA (2 with CABG, 1 with unroofing, and 1 with reimplantation). Both patients with CABG (1 with saphenous vein graft and 1 with right IMA) had early recurrence of symptoms and graft failure. In the present series, the only 2 patients that had recurrent symptoms of chest pain had undergone CABG as treatment for AAOCA.
Reimplantation has also been described with good success, but may be technically more challenging. The buttons may be challenging to form as well as there may be issues with reimplanting at an agreeable angle. Pulmonary artery translocation was described by Gulati and colleagues [15] for treatment of a single coronary ostia. Another option is unroofing of the affected anomalous coronary with or without detachment of the aortic valve commissure [11, 12, 14, 16]. Results using this technique have been good and we feel that unroofing offers a simple technique with safe, reproducible outcomes. Our practice has evolved to utilizing the unroofing technique over CABG whenever technically feasible.
The current practice at the Mayo Clinic is to recommend surgical repair of all patients with ALCA regardless of the presence or absence of symptoms. Management of ARCA has been a subject of debate. Although the association of ARCA with sudden death is not quite as convincing as ALCA, it does exist [2, 3, 6–8]. The practice at the Mayo Clinic has been to thoroughly evaluate these patients with stress testing and to recommend surgical intervention to all symptomatic patients with ARCA. Surgery is also recommended for patients with ARCA and a positive stress testing for patients with very active lifestyles. A nondominant ARCA in a patient without symptoms would be an exception. Unroofing the AAOCA is our preferred surgical treatment with CABG being reserved for patients with concomitant atherosclerotic disease or in patients with anatomy not amenable to unroofing.
This study is a small retrospective study and thus has the inherent limitations of all retrospective studies. The study size is small but it is the largest series to date. Our experience with treating this disease has increased since the year 2000 and thus our follow-up time is short. Longer follow-up would be needed to document the benefits of surgical intervention in this group.
Anomalous aortic origin of a coronary artery from the opposite sinus is a rare, potentially lethal cardiac anomaly. All patients with symptomatic AAOCA and asymptomatic ALCA should undergo repair. Asymptomatic ARCA warrants repair in the face of an abnormal stress test or if lifestyle will be limited. Surgical unroofing of the affected coronary artery can be performed safely with excellent results.
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Discussion |
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Also, I wonder in your title of your talk, if you would consider changing the nomenclature to what I think most people are calling this and that is an anomalous aortic origin of the coronary artery. The reason for this suggestion is that there are on-going prospective studies that are referring to this anatomy as anomalous aortic origin. I think that your paper is important enough to be included in the upcoming discussion that is sure to take place. Thank you.
DR DAVIES: Yes, we would consider changing the name. But the first question. How many were underneath the commissure? Approximately 25% were underneath the commissure. We treated them in a combination of ways. One was taking down the commissure and tacking the commissure back up after unroofing. A couple of the procedures performed more recently were treated with fenestration of the anomalous track. We didn't take down the commissure and just unroofed the portion that was on the opposite side of the commissure.
DR MAVROUDIS: What happened to the one that—this is a really dangerous thing to do in an aortic valve, and I think Jim Jaggers, if he's here, might speak against it—but what happened to those patients where you took the commissure down, did the unroofing and put the commissure back, did those aortic valves work okay?
DR DAVIES: Yes, sir.
DR MAVROUDIS: They did? Good for you.
DR JOHN LAMBERTI (San Diego, CA): Can you tell us what happened to the nine positive stress tests after repair?
DR DAVIES: None of those patients had recurrent symptoms. We do not have a lot of stress echoes postoperatively yet. We recently started performing these more commonly, and all of those have been negative, but it was not done in the earlier portion of the study.
DR RALPH MOSCA (New York, NY): Thank you for a nice presentation of a very interesting topic, which is receiving a good deal of attention lately.
Am I correct in stating that your work-up of patients with anomalous right coronary artery from the left sinus includes lifestyle and stress test results and that these are used in the decision making process regarding surgery? Are the stress tests treadmill tests or stress echos?
DR DAVIES: Yes, sir.
DR MOSCA: I can understand how a positive stress test might be helpful in this lesion. However, do you believe the tests are sensitive and specific enough to play an important part in your treatment algorithm?
DR DAVIES: For the asymptomatic right coronary artery, we do do that. For the symptomatics, we recommend surgery for those. For the asymptomatic we do perform it. I don't have the data to say exactly how many of those were positive versus negative in that group because we only had nine of the total patients that had positive stress test. We do use it. It's not absolute, but we do. Obviously, if it's positive, it helps in our decision-making.
DR MARSHALL JACOBS (Newtown Square, PA): I think it's an important study from your institution and adds a lot to our understanding of this rare anomaly. As you may know, the Congenital Heart Surgeons Society is embarking on creation of a multisocietal registry to look at these patients.
I have two questions. One is related to Dr Mosca's question, and it has to do with my understanding of what you said about postoperative stress tests. This past year authors from the Children's Hospital of Philadelphia published a series wherein they did radionuclide stress perfusion studies after surgery for anomalous aortic origin of a coronary artery. And of the patients who underwent unroofing and repair of anomalous right coronary from the left sinus, several had perfusion defects on the postoperative stress tests despite being asymptomatic. Could you please clarify how many of the patients in your report underwent stress perfusion studies.
My second question concerns the group of patients who were excluded from your analysis. In terms of various hypotheses about risk stratification in these patients, I think we can learn a lot about the ones whose diagnosis isn't discovered until they present in the fifth or sixth decade of life with advanced coronary atherosclerosis. If they have survived to that point, despite the presence of a slit-like orifice, or an intramural segment, or an interarterial course, that may have implications for our understanding of the significance of those features of the disease.
DR DAVIES: The first question, of the patients, again we just now started really looking at stress testing postoperatively; there have only been three patients and they have all been negative. And those all three were of right coronaries.
Interestingly, for our patients, I'm not sure of the referral pattern or what, but we've seen more commonly anomalous rights recently, by at least the 6-to-1 ratio that I showed.
The second question about the 14 that we eliminated, those patients had, when we looked back at them, they had a little bit higher number of recurrent symptoms. It was not statistically significant because the numbers are so small, but it was a higher number of recurrent symptoms for those patients. It was unclear whether or not those recurrent symptoms were more related to their atherosclerotic disease or if it was related to the unroofing or it wasn't maturing of the IMAs [internal mammary arteries] or the CABG [coronary artery bypass grafting] patients. But we felt that it was more likely more related to the fact that they had progression of their atherosclerotic disease.
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This article has been cited by other articles:
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H. M. Burkhart Reply Ann. Thorac. Surg., April 1, 2010; 89(4): 1341 - 1342. [Full Text] [PDF]
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