Anomalous Left Coronary Artery from the Pulmonary Arteryin an Adult

2012 ◽  
Vol 15 (5) ◽  
pp. 284
Author(s):  
John D. Vossler ◽  
Daniel W. Entrikin ◽  
Michael D. Quartermain ◽  
Neal D. Kon ◽  
Ross M. Ungerleider ◽  
...  

Anomalous left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital heart defect that usually presents before the age of 1 year. Several surgical options exist for the correction of ALCAPA; however, debate continues regarding the optimal repair technique in adult populations. We report the case of successful surgical repair of ALCAPA with a direct aortic implantation technique in a 44-year-old mother of 4 children.

2020 ◽  
Vol 30 (3) ◽  
pp. 362-368
Author(s):  
Amr A. Matoq ◽  
Takeshi Tsuda

AbstractBackground:Anomalous left coronary artery from the pulmonary artery is a rare congenital heart disease (CHD) with diverse clinical presentations despite the same anatomy. Factors determining this heterogeneous presentation are not well understood.Method and Results:We retrospectively investigated 14 patients (12 females) who underwent surgical repair of anomalous left coronary artery from the pulmonary artery. These patients were divided into three groups based upon the severity of initial presentation: (1) severe, life-threatening condition (n = 5), (2) mild-to-moderate distress (n = 6), and (3) asymptomatic (n = 3). All patients presented with left ventricular dilation and retrograde flow in left coronary artery by echocardiogram. Eight patients in (1) and (2) presented with severe left ventricular dysfunction. All but one showed abnormal ECG consistent with myocardial ischemia or infarction. Asymptomatic patients had preserved left ventricular systolic function despite ischemic findings on ECG. In 13 patients after surgical repair, all but one normalised left ventricular geometry and systolic function, suggesting nearly full myocardial recovery upon improvement of myocardial perfusion; 8 patients had residual echogenic papillary muscle with variable degree of mitral regurgitation.ConclusionsEvidence of myocardial ischemic injury was present in all patients with anomalous left coronary artery from the pulmonary artery regardless of their initial presentation. Retrograde flow in left coronary artery, implying collateral vessel development from right coronary artery to left coronary artery, was noted in all patients, yet only few patients had preserved systolic function at the time of diagnosis. The balance between effective myocardial perfusion and a deleterious fistulous flow provided by these collaterals and the simultaneous haemodynamic status are what determine the clinical diversity of anomalous left coronary artery from the pulmonary artery.


2015 ◽  
Vol 26 (4) ◽  
pp. 802-804
Author(s):  
Xuecun Liang ◽  
Lin Wu

AbstractIn this study, we describe a very rare case of anomalous left coronary artery arising from the right pulmonary artery in association with coarctation of the aorta. A 3-month-old boy presented with refractory congestive heart failure since 20 days after birth. The initial echocardiography suggested the diagnosis of left coronary artery-to-right pulmonary artery fistula associated with coarctation; however, selective coronary angiography demonstrated the rare anomaly of the left coronary artery arising from the right pulmonary artery. Subsequently, he underwent successful transcatheter balloon angioplasty for aortic coarctation and surgical repair of left coronary artery re-implantation.


2013 ◽  
Vol 16 (4) ◽  
pp. 210 ◽  
Author(s):  
Sachin Talwar ◽  
Aandrei Jivendra Jha ◽  
Shiv Kumar Choudhary ◽  
Saurabh Kumar Gupta ◽  
Balram Airan

Between January 2002 and December 2012, five patients (4 female) underwent corrective surgery for anomalous left coronary artery from pulmonary artery (ALCAPA). They were older than 1 year (range, 3-56 years). One of the 2 patients younger than 10 years had presented with congestive heart failure, and the other had experienced repeated episodes of lower respiratory tract infection since childhood. Of the remaining 3 adult patients, 2 had experienced angina with effort, and 1 patient had had repeated respiratory tract infections since childhood, with mild dyspnea on effort of New York Heart Association (NYHA) class II. Three patients had the anomalous left coronary artery implanted directly into the ascending aorta via coronary-button transfer, and 2 patients underwent coronary artery bypass with obliteration of the left main ostium. Two patients underwent concomitant mitral valve repair procedures, and 1 patient underwent direct closure of a perimembranous ventricular septal defect. Four patients survived the surgery, and 1 patient died because of a persistently low cardiac output. Follow-up times ranged from 3 months to 4 years. All survivors are in NYHA class I and have left ventricular ejection fractions of 45% to 60%, with moderate (n = 1), mild (n = 1), or no (n = 2) mitral insufficiency. We conclude that a few naturally selected patients with ALCAPA do survive beyond infancy and can undergo establishment of 2 coronary systems with satisfactory results.


2020 ◽  
Vol 4 (3) ◽  
pp. 1-5
Author(s):  
Madelien V Regeer ◽  
Olga Bondarenko ◽  
Katja Zeppenfeld ◽  
Anastasia D Egorova

Abstract Background Anomalous left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital disorder resulting in ischaemia and myocardial infarction which can act as a potential substrate for life-threatening arrhythmias and sudden cardiac death. Case summary A 19-year-old man was admitted to the hospital after successful resuscitation from an out-of-hospital cardiac arrest (OHCA) due to ventricular fibrillation occurring during jogging. In the diagnostic work-up of the OHCA, computed tomography identified an ALCAPA. The patient was referred to our tertiary hospital for surgical correction. Direct reimplantation of the left coronary artery in the aorta was performed. During follow-up, 24-h electrocardiogram revealed short episodes of non-sustained ventricular tachycardia (VT). The magnetic resonance imaging at initial admission showed focal wall thinning and transmural late gadolinium enhancement consistent with a previous anterolateral myocardial infarction. Therefore, the aetiology of the OHCA could be due to a scar-related mechanism and not necessarily due to a reversible cause and an implantable cardioverter-defibrillator (ICD) was considered indicated. Given the young age and the lower complication rates, a subcutaneous device was preferred over a transvenous ICD. However, as a subcutaneous ICD (S-ICD) lacks the possibility of anti-tachycardia pacing, programmed electrical stimulation (PES) was performed to test for inducibility of monomorphic, re-entrant VT. After a negative PES, an S-ICD was implanted. Discussion ALCAPA is a potential cause of OHCA in young patients. Some of these patients keep an irreversible substrate for ventricular arrhythmias despite full surgical revascularization and might be candidates for (subcutaneous) ICD implantation.


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