Anomalous origin of left coronary artery from pulmonary artery: A rare cause of myocardial infarction in children

Lardhi, Amer A.

doi:10.4103/1319-1683.74319

Cited by 29 publications

(22 citation statements)

References 20 publications

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“…Embryologically, the anomaly arises from either abnormal septation of the aorta and the pulmonary artery or from persistence of aortic buds that form the coronary arteries [4].…”

Section: Discussionmentioning

confidence: 99%

Adult ALCAPA Syndrome-Anomalous Origin of Left Coronary Artery from Pulmonary Artery: A Rare Coronary Artery Anomaly Diagnosed by 256-Slice Computed Tomography

Taori¹,

Dhakate²,

Hatgaonkar³

et al. 2015

OALib

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We are presenting a rare case of anomalous origin of left coronary artery from the pulmonary artery (ALCAPA) in an adult patient. The patient underwent coronary CT angiography for exertional dyspnoea, suspected as coronary artery disease; however there were no atherosclerotic plaques. There was anomalous origin of left coronary artery from pulmonary artery. Conventional catheter angiography confirmed the origin of left coronary artery from pulmonary artery, which was retrogradely filling through right coronary artery.

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“…Embryologically, the anomaly arises from either abnormal septation of the aorta and the pulmonary artery or from persistence of aortic buds that form the coronary arteries [4].…”

Section: Discussionmentioning

confidence: 99%

Adult ALCAPA Syndrome-Anomalous Origin of Left Coronary Artery from Pulmonary Artery: A Rare Coronary Artery Anomaly Diagnosed by 256-Slice Computed Tomography

Taori¹,

Dhakate²,

Hatgaonkar³

et al. 2015

OALib

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“…1 It was first described in 1886, but Bland, Garland and White described in 1933 the landmark case describing the clinical features linked with ALCAPA; hence it is also known as Bland-White-Garland syndrome. 2,3 There are two types of ALCAPA syndrome: the adult and the infant type. The infant type presents with myocardial ischaemia or infarction and/or cardiac failure, and has a mortality rate of 90% within the first year of life.…”

Section: Discussionmentioning

confidence: 99%

“…4 Embryologically, the anomaly arises from either abnormal septation of the aorta and the pulmonary artery, or from persistence of aortic buds that form the coronary arteries. 2 In the neonatal period, the baby is asymptomatic as there is anterograde flow of desaturated blood from the pulmonary artery to the left coronary artery. As pulmonary arterial pressure drops, the combination of low flow and desaturated blood causes myocardial ischaemia, especially during exertion.…”

Section: Discussionmentioning

confidence: 99%

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ALCAPA: The Al Capone of coronary artery anomalies

Ismail

2012

S. Afr. j. radiol.

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A 12-month-old infant presented with tachycardia and respiratory distress. On clinical evaluation, the child was in cardiac failure. There were no clinical features of infection. Haematological studies were normal. The electrocardiogram (ECG) demonstrated septal ischaemic changes. The chest radiograph (Fig. 1) demonstrated gross cardiomegaly involving mainly the ventricles and right atrium. There was peri-hilar alveolar opacification and a left pleural effusion. There was a positive silhouette sign at the right cardiac border, indicating right middle lobe consolidation. These findings were due to pulmonary oedema or infection. The differential diagnosis at this stage was dilated cardiomyopathy, viral myocarditis, mitral valve disease or anomalous left coronary artery originating from the pulmonary artery (ALCAPA). Echocardiography excluded cardiomyopathy and mitral valve disease. Coronary angiography confirmed ALCAPA (Figs 2 and 3). The patient was evaluated for surgery. DiscussionAnomalous left coronary artery originating from the pulmonary artery (ALCAPA) is a rare coronary artery anomaly that affects 1 in 300 000 live births.1 It was first described in 1886, but Bland, Garland and White described in 1933 the landmark case describing the clinical features linked with ALCAPA; hence it is also known as Bland-White-Garland syndrome. 2,3There are two types of ALCAPA syndrome: the adult and the infant type. The infant type presents with myocardial ischaemia or infarction and/or cardiac failure, and has a mortality rate of 90% within the first year of life.1 Infants may also present with paroxysms of irritability which correlate with episodes of angina.4 ALCAPA can occur in isolation or in conjunction with other lesions such as atrial septal defect and ventricular septal defect. 4 Embryologically, the anomaly arises from either abnormal septation of the aorta and the pulmonary artery, or from persistence of aortic buds that form the coronary arteries.2 In the neonatal period, the baby is asymptomatic as there is anterograde flow of desaturated blood from the pulmonary artery to the left coronary artery. As pulmonary arterial pressure drops, the combination of low flow and desaturated blood causes myocardial ischaemia, especially during exertion. Collateral vessels develop between the right and left coronary arteries. Further decreases in pulmonary arterial pressure result in reversal of flow, as the left coronary artery drains from the right coronary artery, through collaterals, into the pulmonary artery. This is known as myocardial steal; hence the nickname of Al Capone of coronary vessels. 2,4The steal phenomenon causes ischaemia or infarction of the anterolateral LV wall. The electrocardiogram findings include a QR pattern followed by inverted T waves seen on leads I and aVL. The left ventricular surface leads (V 5 -V 6 ) may also show deep Q waves and exhibit elevated ST segments and inverted T waves.2 Patients who survive to adulthood have a good collateral network with large-calibre left and right coronary art...

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“…This is known as myocardial steal or coronary steal. 12,13 In the infants, the chief symptom was irritability elicited by only slight effort, such as feeding, with signs of poor peripheral perfusion. These features started within 2 months of birth, coinciding with substantial reduction in pulmonary vascular resistance that resulted in coronary steal from the anterolateral aspect of the LV.…”

Section: Discussionmentioning

confidence: 99%

Anomalous Origin of Left Coronary Artery from the Pulmonary Artery (ALCAPA) in an Infant - A Case Report

Jabbar

Hussain

Rima

et al. 2017

Bangladesh J Child Health

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IntroductionAnomalous origin of the left coronary artery (LCA) from the pulmonary artery (abnormal left coronary artery from pulmonary artery = ALCAPA) or BladeWhite-Garland syndrome is a rare, but serious congenital cardiac malformation. ALCAPA represents one of the most common causes of myocardial ischaemia and infarction in children. If left untreated, the mortality rate is upto 90% within the first year of life. ALCAPA accounts for 0.25-0.50% of all congenital heart diseases. 1 Although the anomalous origin of the coronary arteries that arise from the pulmonary artery was first described in1886. 2 It was not until 1933 when Bland et al 3 described the first clinical features with an autopsy finding of anomalous left coronary artery arise from the pulmonary artery (ALCAPA). The anomaly has thus been called the Bland-White-Garland Syndrome. 3,4 A coronary artery anomaly may involve an abnormal number, origin and/ or course, termination or structure of the coronary arteries. 5 The left main coronary artery normally arises from the left coronary sinus of valsalva. Instead of arising from the left aortic sinus, it may arises from the right sinus of valsalva or the proximal right coronary artery, from the non-coronary (posterior) aortic sinus,

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Anomalous origin of left coronary artery from pulmonary artery: A rare cause of myocardial infarction in children

Cited by 29 publications

References 20 publications

Adult ALCAPA Syndrome-Anomalous Origin of Left Coronary Artery from Pulmonary Artery: A Rare Coronary Artery Anomaly Diagnosed by 256-Slice Computed Tomography

Adult ALCAPA Syndrome-Anomalous Origin of Left Coronary Artery from Pulmonary Artery: A Rare Coronary Artery Anomaly Diagnosed by 256-Slice Computed Tomography

ALCAPA: The Al Capone of coronary artery anomalies

Anomalous Origin of Left Coronary Artery from the Pulmonary Artery (ALCAPA) in an Infant - A Case Report

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