Eur J Cardiothorac Surg 1999;15:539-541
© 1999 Elsevier Science NL
Malignant ventricular arrhythmias revealing anomalous origin of the left coronary artery from the pulmonary artery in two adults
Jean-Marc Frapiera,
Florence Leclercqb,
Marc Bodinoa,
Paul-André Chaptala
a Department of Cardiovascular Surgery, Arnaud de Villeneuve University Hospital, Montpellier, France
b Department of Cardiology, Arnaud de Villeneuve University Hospital, 34295 Montpellier Cedex 5, France
Received 14 September 1998;
received in revised form 28 December 1998;
accepted 12 January 1999.
Corresponding author. Tel.: +33-4-6733-6272; fax: +33-4-6733-6275; e-mail: pa-chaptal@chu-montpellier.fr
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Abstract
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We report two cases of anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA), revealed by malignant ventricular arrythmias in adult patients. A two coronary system was re-established in both patients, and cryotherapy was performed on one of the patients who, in addition, presented ventricular aneurysm triggering ventricular tachycardia.
Key Words: Anomalous origin of the left coronary artery from the pulmonary artery • Ventricular tachycardia • Ventricular fibrillation • Cryotherapy
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Introduction
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Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rarely detected congenital defect in adults. Sudden cardiac death is a major clinical presentation of ALCAPA in adults and is presumed to be caused by malignant ventricular arrhythmias. However, electrocardiographic documentation of such arrhythmias remains very rare
[1]
[2]
[3]
[4]
[5]
[6]. Here, we report two such cases. Further, in addition to the re-establishment of a two coronary system, cryotherapy against ventricular tachycardia(VT) was performed on one of the patients.
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Case reports
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Case 1
A 38-year-old female was admitted following resuscitation from cardiac arrest. During reanimation by the mobile care team, she presented polymorphic ventricular tachycardia (VT) at 280 bpm which degenerated in ventricular fibrillation (VF). A physical examination revealed a 1/6 systolic murmur in the left superior sternal border. An electrocardiogram showed sinus rhythm, short P–R interval (10 ms), left axis deviation, poor R wave progression in leads V1 to V2 with a QR aspect in the V3 lead. An echocardiogram revealed a slight dilation of the left ventricle (diastolic diameter 57 mm), and mild global hypokinesia. During coronary angiography, contrast medium injected into a huge right coronary artery filled the left anterior descending and circumflex coronary arteries with retrograde opacification of the pulmonary artery. Left ventriculography showed a 0.62 ejection fraction without segmental dyskinesia. An electrophysiologic study (EPS) failed to induce a sustained VT. The patient underwent surgical correction under cardiopulmonary bypass with tepid temperature and cold blood cardioplegia. Due to a short left main coronary artery, attempt at direct aortic reimplantation of the ALCAPA located on the left posterior sinus of the pulmonary artery failed, and closure of the left main and mammary artery anastomosis to the left anterior descending coronary artery were performed. Following recovery, a maximal-exercise stress test failed to reveal evidence of ischemia and AICD implantation was not considered. During a 13-month follow-up period she remained asymptomatic.
Case 2
A 47-year-old woman had a syncopal episode. She was found by the mobile care team in monomorphic sustained VT at 250 bpm, with right bundle branch block like morphology and left axis deviation, compatible with left ventricular origin. Although previously asymptomatic, she had been treated with verapamil for ventricular premature beats during the 3 years preceding her syncopal episode. Upon admission, an electrocardiogram showed sinus rhythm, left axis deviation, and anteroseptal sequelae. Left cardiac catheterization revealed an ALCAPA (
Fig. 1
), a small high anterolateral dyskinesia in the RAO projection of the ventriculogram (
Fig. 2
), and a 0.70 ejection fraction. A sustained VT was inducible by an EPS. The patient underwent surgical correction by cardiopulmonary bypass under tepid temperature and cold blood cardioplegia. A direct aortic reimplantation using the Tashiro procedure
[7] was performed, along with an encircling cryotherapy of the aneurysm border zone through a short left ventriculotomy centred in the scar using the Guiraudon technique
[8]. Recovery was uneventful and postoperative EPS was negative. After 2 months, without medication, maximal exercise stress tests failed to reveal evidence of ischemia and AICD implantation was not considered. However, two bursts of premature ventricular beats (up to 5 beats) were recorded on holter ECG; a 100 mg daily regimen of atenolol was prescribed. A subsequent holter ECG, under atenolol, was normal. The patient remained asymptomatic during a 11-month follow-up period.
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Fig. 1. Coronary angiogram: the contrast medium injected into the right coronary artery filled the left anterior descending and circumflex coronary arteries with retrograde opacification of the pulmonary artery.
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Discussion
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The discovery of ALCAPA in adults as a rare and sudden cardiac death is a major clinical presentation
[5] and is due to malignant ventricular arrhythmias. These arrhythmias are of acute ischemic origin and the restoration of a dual coronary system, by preventing further ischemia of the left ventricle, results in angina cessation, normalization of stress ECG and the disappearance of arrhythmia
[6]. Depending upon the anatomical location of the ostium of the ALCAPA, several procedures are suggested to achieve this goal in adults: saphenous vein or mammary artery grafting (employed in our first case); reimplantation into the aortic root, directly or with the help of a pulmonary flap
[7] (as performed in our second case); or the construction of a transpulmonary tunnel as described by Takeuchi. Subsequent improvement of left ventricular function is common, however, this improved function may eventually be impaired by scar tissue. Moreover, after a myocardial infarction, left ventricular scar tissue may become arrhythmogenic and trigger VT. The use of aneurysmectomy, as a method of surgical correction of an ALCAPA in order to treat heart failure, has been reported for adults and infants
[9]. However, the use of a direct antitachycardia procedure has been reported in only one case, in which a child underwent map-guided endocardial resection associated with mammary artery grafting
[10]. In our second case, a preoperative evaluation showed a well defined anterolateral aneurysm. As an EPS induced sustained VT, we elected to perform the Guiraudon procedure
[8] associated with an aortic reimplantation of the left main coronary artery.
Since ischemia associated with ALCAPA, may induce malignant arrhythmias, the detection of an aneurysmal scar should prompt further exploration in order to assess the value of an additional procedure to the restoration of a dual coronary system.
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References
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Abstract
Introduction
