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Ann Thorac Surg 2008;85:2135-2136. doi:10.1016/j.athoracsur.2007.07.006
© 2008 The Society of Thoracic Surgeons
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William C. DeVries
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Case Reports

Repair of Anomalous Origin of Right Coronary Artery From the Left Sinus of Valsalva

Houman Tavaf-Motamen, MD*, Sean P. Bannister, PA-C, Philip C. Corcoran, MD, Robert W. Stewart, MD, Charles R. Mulligan, MD, William C. DeVries, MD

Division of Cardiothoracic Surgery, Walter Reed Army Medical Center, Washington, DC

Accepted for publication July 5, 2007.

* Address correspondence to Dr Tavaf-Motamen, Walter Reed Army Medical Center, Cardiothoracic Surgery Division, 6900 Georgia Ave, Washington, DC 20303 (Email: houman.tavaf-motamen{at}na.amedd.army.mil).


   Abstract
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 Abstract
Introduction
 Case Reports
 Comment
 References
 
Anomalous origin of the right coronary artery from the left sinus of Valsalva is a rare congenital anomaly but a relatively frequent cause of sudden death in the young. The medical records of 4 consecutive patients with this anomaly were retrospectively reviewed. The first 2 patients underwent coronary artery bypass and had early graft failure. The next 2 patients underwent coronary reimplantation and unroofing and have done well. This limited series suggests that bypass procedures used to treat anomalous origin of the right coronary artery from the left sinus of Valsalva are prone to early graft failure.


   Introduction
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 Abstract
 Introduction
Case Reports
 Comment
 References
 
Anomalous origin of the right coronary artery (RCA) from the left sinus of Valsalva is a rare congenital anomaly but a common cause of sudden death in the young. We describe the outcomes of 4 consecutive patients seen with this anomaly treated with four different surgical techniques at Walter Reed Army Medical Center between March 2004 and January 2007.


   Case Reports
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Patient 1
This 56-year-old man presented with exertional angina. A nuclear perfusion scan showed no evidence of ischemia. Coronary angiography revealed an anomalous RCA arising from the left sinus of Valsalva. This patient underwent an on-pump bypass, which used a saphenous vein to the distal RCA. The proximal RCA was not ligated. Six months postoperatively, the patient noted return of his angina. Stress echocardiography revealed inferolateral hypokinesis, and repeat cardiac catheterization revealed flush occlusion of the graft. Patient was offered reoperation but declined.

Patient 2
This man presented at 33 years old with a 1-year history of exertional angina. Cardiac catheterization and computed tomography (CT) angiogram (CTA) revealed anomalous origin of the RCA from the left sinus of Valsalva. The patient underwent an off-pump right internal mammary artery (RIMA) bypass to the distal RCA. The proximal RCA was not ligated. Three months postoperatively, the patient experienced recurrent angina. Cardiac catheterization revealed a patent but diminutive RIMA to RCA graft with minimal flow.

Patient 3
This 51-year-old man sustained an inferior wall myocardial infarction that was initially treated with thrombolytics. Coronary angiography revealed a slit-like ostium of the RCA, with anomalous origin from the left sinus of Valsalva. Results of echocardiogram and exercise treadmill test were both normal. This patient was noted in the operating room to have a superficial and ectatic proximal RCA. The RCA was divided and reimplanted by using an end-to-side aortocoronary anastomosis to the right sinus of Valsalva. Postoperatively, the patient has had a normal result on an exercise myocardial perfusion scan and stress echocardiography and remains asymptomatic 2 years later.

Patient 4
This 35-year-old man presented to the emergency department with chest pain. The result of patient's electrocardiogram was normal, but he had an elevated troponin level. Exercise stress test revealed ST depression in the inferior leads. Subsequent cardiac catheterization revealed an RCA with an anomalous origin from the left coronary cusp and a slit-like ostium. The exercise myocardial perfusion scan was remarkable for inferior reversible ischemia. A CTA confirmed the anomalous origin of the RCA. The patient underwent an unroofing of the intramural segment of the proximal RCA by way of a transverse aortotomy. Postoperative CTA revealed an anterior translocation of the RCA ostium with relief of the ostial obstruction. The myocardial perfusion scan was without evidence of ischemia, and the patient remains asymptomatic 6 months postoperatively.


   Comment
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References
 
Anomalous RCA from the left sinus of Valsalva is a rare congenital anomaly but a relatively frequent cause of sudden death in the young. The mechanism of myocardial ischemia and sudden death in these patients is not completely elucidated, but proposed causes include:

1 Ostial obstruction due to slit-like coronary orifice;
2 Compression of RCA between aorta and pulmonary artery;
3 Stretching of the RCA with aortic/pulmonary artery distention;
4 Coronary angulation with aortic/pulmonary artery distention [1].

As opposed to atherosclerotic coronary artery disease, the nature of these obstructions is dynamic. Symptoms often occur only at maximal exertion, and routine screening tests, such as myocardial perfusion scans, are frequently nondiagnostic.

Initially, it was thought that anomalous origin of the RCA from the left sinus of Valsalva was clinically benign and required no specific treatment. With the association with sudden death, several approaches were advocated to mitigate this risk. These repairs can be classified as either bypass procedures or procedures designed to establish normal coronary anatomy. In this series, two bypass procedures had early failure and two procedures designed to establish normal coronary anatomy were successful.

Anecdotal evidence in the literature suggests that bypass procedures for this condition are prone to failure. Fedoruk and colleagues [2] reported 4 patients who underwent unroofing procedures and 1 patient who underwent an RCA reimplantation with good results. Two of the patients in this series had previously undergone RIMA-to-RCA bypass procedures that resulted in early graft failure [2]. Reul and colleagues [3] report a patient with graft occlusion who 1 month previously had undergone RIMA-to-anomalous RCA bypass at another institution.

The association with early graft failure may be related to the dynamic nature of the obstructive process. Often, the impediment to coronary perfusion occurs only during high levels of exertion. At lower levels of exertion in a patient with an anomalous RCA who has undergone a bypass graft, most of the blood flow will occur down the native RCA and not through the bypass conduit. There is evidence that the competitive flow phenomenon can be mitigated by ligation of the RCA proximal to the bypass graft [3]. We have been reticent to ligate the RCA in young patients because of concern that this would render them graft-dependant and prone to infarction if the graft fails.

Although unroofing the intramural aspect of the RCA is relatively easy and generally has good results, it can undermine the aortic valve commissure. Romp and colleagues [4] describe 7 patients with anomalous left main and 2 patients with anomalous RCA in whom a modified unroofing procedure was used. Severe aortic insufficiency developed in 1 patient who underwent unroofing, and aortic valve replacement was required [4].

Reimplantation of the RCA is also well described [5]. The advantage of this technique compared with unroofing is that it leaves the aortic valve unmolested. Technical considerations in its execution include adequate mobilization to ensure a tension-free anastomosis as well as prevention of kinking or torsion or both.

This limited series suggests that bypass procedures without RCA ligation are prone to early failure. Our current operative strategy is to perform transverse aortotomy after arresting the heart. We proceed with an unroofing procedure if inspection shows the intramural aspect of the RCA is favorably oriented in relation to the aortic valve commissure. On the other hand, if the inspection indicates that unroofing the RCA will undermine the aortic valve, we recommend reimplantation of the coronary through a separate punch hole in the right sinus of Valsalva. Bypass procedures should be reserved for patients with concomitant obstructive atherosclerotic coronary artery disease unless one also plans on ligating the proximal RCA.


   References
Top
 Abstract
 Introduction
 Case Reports
 Comment
 References
 

  1. Taylor A, Rogan K, Virmani R. Sudden cardiac death associated with isolated congenital coronary artery anomalies J Am Coll Cardiol 1992;20:64-67.
  2. Fedoruk L, Kern J, Peeler B, Kron I. Anomalous origin of the right coronary artery: right internal thoracic artery to right coronary artery bypass is not the answer J Thorac Cardiovasc Surg 2007;133:456-460.[Abstract/Free Full Text]
  3. Reul R, Cooley D, Hallman G. Surgical treatment of coronary artery anomalies: report of a 37 1/2-year experience at the Texas Heart Institute Tex Heart Inst J 2002;29:299-307.[Medline]
  4. Romp R, Herlong JR, Landolfo C, et al. Outcome of unroofing procedure for repair of anomalous aortic origin of the left or right coronary artery Ann Thorac Surg 2003;76:589-596.[Abstract/Free Full Text]
  5. Rogers S, Leacche M, Mihaljevic T, Rawn J, Byrne J. Surgery for anomalous origin of the right coronary artery from the left aortic sinus Ann Thorac Surg 2004;78:1829-1831.[Abstract/Free Full Text]



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This Article
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Houman Tavaf-Motamen
Philip C. Corcoran
Robert W. Stewart
Charles R. Mulligan
William C. DeVries
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Right arrow Congenital - acyanotic

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