Ann Thorac Surg 2007;83:1175-1177
© 2007 The Society of Thoracic Surgeons
Case Reports
Anomalous Left Anterior Descending Coronary Artery From Pulmonary Artery With Ventricular Septal Defect
Thomas Stephen, MCh,
Korah T. Kuruvila, MCh,
Madhu Andrew Philip, MCh,
Vinayak Shukla, MCh, DNB,
Roy John Korula, MCh, FACS*
Department of Cardiothoracic Surgery, Unit II, Christian Medical College, Vellore, India
Accepted for publication September 12, 2006.
* Address correspondence to Prof Korula, Department of Cardiothoracic Surgery Unit II, Christian Medical College, Vellore 632 004, India (Email: roykorula{at}hotmail.com).
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Abstract
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Anomalous origin of the left anterior descending coronary artery with associated congenital defects is very rare. An angiogram of a 47-year-old woman admitted for a ventricular septal defect closure revealed an anomalous left anterior descending coronary artery arising from the left posterior sinus of the pulmonary artery. During the surgical procedure, the origin of the left anterior descending coronary artery was closed with pledgetted polypropylene sutures through the pulmonary artery. The ventricular septal defect was closed with a patch through the right atrium, and the left anterior descending coronary artery was bypassed with the left internal mammary artery.
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Introduction
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Anomalous origin of the left anterior descending coronary artery (LAD) from the pulmonary artery is rare. The reported incidence varies from 0.26% to 0.46% of congenital heart disease. Its association with other congenital heart defects is even rarer. We report a patient with anomalous origin of the LAD from the pulmonary artery with ventricular septal defect.
A 47-year-old woman was admitted with complaints of exertional dyspnea and palpitations (New York Heart Association [NYHA] class II) and occasional episodes of syncopal attacks for 18 years. During her initial visits in 1987, she was diagnosed on echocardiogram to have idiopathic pulmonary artery dilatation with pulmonary artery hypertension and moderate tricuspid regurgitation. She was treated conservatively for this. Subsequently in 1994, during her fourth pregnancy, a repeat echocardiogram revealed a small subaortic ventricular septal defect with moderate pulmonary artery hypertension (pulmonary artery systolic pressures, 55 mm Hg) and moderately severe tricuspid regurgitation. She refused further treatment because of financial constraints.
Seven years later, low-grade fever developed in association with anorexia and weight loss. Laboratory investigations and an echocardiogram confirmed infective endocarditis with vegetations on the tricuspid valve. She was treated medically and was advised to have surgical closure of the ventricular septal defect at the earliest after cardiac catheterization and coronary angiogram.
On admission, there was a systolic thrill and a grade 4/6 pansystolic murmur in the left parasternal area. The second heart sound was variably split with a loud pulmonary component. There was no evidence of ischemia, infarction, or ventricular hypertrophy on electrocardiogram. The result of chest roentgenogram was normal. Echocardiogram revealed a small, 5-mm subaortic ventricular septal defect with moderate pulmonary hypertension and mild tricuspid regurgitation. The left ventricular ejection fraction was normal (0.58).
Her coronary angiogram (Fig 1) revealed an absent left main coronary artery. The circumflex coronary artery, which was arising from the left aortic sinus, was dominant and ectatic. The LAD was also dilated and ectatic and was seen filling from left to left collaterals and was draining into the left posterior sinus of the pulmonary artery. The right coronary artery was small, not dominant, and normal. The left ventriculogram showed a small subaortic ventricular septal defect with a left-to-right shunt.
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Fig 1. Selective coronary angiogram of left circumflex artery in 90-degree dead lateral view shows opacification of LAD artery draining into PA. (AO = Aorta; CX = circumflex artery; LAD = left anterior descending artery; PA = pulmonary artery.)
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The left internal mammary artery (LIMA) was harvested. Under routine cardiopulmonary bypass with bicaval cannulation, cold antegrade blood cardioplegic arrest was established after the aorta and pulmonary artery were clamped. A transverse incision was made on the pulmonary artery. The origin of the LAD was visualized. This was closed with a 5-0 pledgetted polypropylene suture. The ventricular septal defect was closed with a Dacron (DuPont, Wilmington, DE) patch through the right atrium. A large hole in the anterior leaflet of the tricuspid valve was closed with 5-0 polypropylene suture. There were no vegetations on the tricuspid valve. The LIMA was anastomosed to the proximal LAD.
The patient’s postoperative period was uneventful. A follow-up angiogram 3 months later showed that the LIMA anastomosis to the LAD was functioning well, and there was good filling of the proximal, mid, and distal LAD. The LAD, circumflex, and right coronary arteries were unchanged in caliber, but the collaterals from the circumflex arteries were no longer visualized. She had NYHA class I symptoms and good exercise tolerance.
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Comment
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An anomalous LAD arising from pulmonary artery with associated congenital defects has been reported previously only in two cases. Both were pediatric patients and had tetralogy of Fallot as the associated defect. One patient underwent translocation to the aorta with the use of an autogenous saphenous vein graft [1], and the other underwent Takeuchi repair [2] apart from respective tetralogy repair. Our case is the third one and occurred in an adult patient.
Most patients with anomalous origin of LAD from the pulmonary artery present with symptoms as adults [3]. This late presentation in this patient may be because of the dual arterial supply and sufficient collateral circulation [4]. In symptomatic patients reported earlier, angina was the main symptom, and associated ischemic changes could be shown in the electrocardiogram. Our patient had neither angina nor ischemic changes. This could be explained by two reasons:
- 1 Because of the presence of a subaortic ventricular septal defect, she had moderate pulmonary hypertension that prevented a gross "coronary steal effect" from the LAD, which has been thought to be a cause for myocardial ischemia [5]; and
- 2 Because of dual coronary artery supply and sufficient collateral circulation.
Surgical establishment of a dual coronary artery system is the treatment of choice [6]. Intrapulmonary tunnel repair and coronary artery reimplantation are the common procedures. The tunnel repair has the risk of tunnel occlusion and acquired supravalvular pulmonary stenosis [6]. Coronary reimplantation of the LAD arising from the posterior left sinus (nonfacing) of the pulmonary artery might cause excessive tension to a aortocoronary anastomoses, resulting in an increased incidence of stenosis and obstruction [6]. In our patient, the LAD was arising from the nonfacing sinus of pulmonary artery.
Turley and colleagues [7] devised a tubular reconstruction technique in children for an anomalous coronary artery arising from left posterior sinus of pulmonary artery, but this is a time-consuming and technically difficult procedure. In adults, a much simpler revascularization procedure of the LIMA-to-LAD can be done with ease. Robert and colleagues [8] reported this for an anomalous left coronary artery from pulmonary artery repair, and it has proven to be the best in terms of long-term patency, freedom from recurrent angina, and cardiac events.
In conclusion, patients with a dual coronary artery supply and only one major coronary artery arising from the pulmonary artery usually present in adulthood with subtle symptoms. Internal mammary artery anastomoses is a good option. This is technically easy and less time-consuming, with excellent functional outcome.
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References
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- Wilcox WD, Hagler DJ, Lie JT, Danielson GK, Danielson GK, Smith HC, Fulton RE. Anomalous origin of left coronary artery from pulmonary artery in association with intra cardiac lesionsReport of two cases. J Thorac Cardiovasc Surg 1979;78:12-18.[Abstract]
- Yamaguchi M, Tsukube T, Hosokawa Y, et al. Pulmonary origin of left anterior descending coronary artery in tetralogy of Fallot Ann Thorac Surg 1991;52:310-312.[Abstract]
- Roberts WC, Robinowitz M. Anomalous origin of the left anterior descending coronary artery from the pulmonary trunk with origin of the right and left circumflex coronary arteries from aorta Am J Cardiol 1984;54:1381-1383.[Medline]
- Probst P, Pachinger O, Koller H, Niederberger N, Kaindl F. Origin of anterior descending branch of left coronary artery from pulmonary trunk Br Heart J 1976;38:523-525.[Free Full Text]
- Evans JJ, Phillips JF. Origin of the left anterior descending coronary artery from the pulmonary artery3 year angiographic follow-up after saphenous vein bypass graft and proximal ligation. J Am Coll Cardiol 1984;3:219-224.[Abstract]
- Barth MJ, Allen BS, Gulecyuz M, Chiemmongkoltip P, Cuneo B, Ilbawi MN. Experience with an alternative technique for the management of anomalous left coronary artery from the pulmonary artery Ann Thorac Surg 2003;76:1429-1434.[Abstract/Free Full Text]
- Turley K, Szarnicki RJ, Flachsbart KD, Richter RC, Popper RW, Tarnoff H. Aortic implantation is possible in all cases of anomalous origin of the left coronary artery from the pulmonary artery Ann Thorac Surg 1995;60:84-89.[Abstract/Free Full Text]
- Chan RKM, Hare DL, Buxton BF. Anomalous left coronary artery arising from the pulmonary artery in an adult: treatment by internal mammary artery grafting J Thorac Cardiovasc Surg 1995;109:393-394.[Free Full Text]
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Abstract
Introduction
