CARDIOVASCULAR JOURNAL OF AFRICA • Vol 23, No 8, September 2012
AFRICA
e15
Case Report
A young patient with coronary artery anomaly, whose
left anterior descending artery originated from the
pulmonary artery, underwent cardiac arrest
TAYFUN SAHİN, SERDAR BOZYEL, ESER ACAR, ULAS BİLDİRİCİ, SADAN YAVUZ, OZGUR BARIS,
ERTAN URAL, DİLEK URAL
Abstract
A rare congenital anomaly of the coronary arteries, in which
the left coronary arterial system starts from the arteria
pulmonalis, is known as Bland–White–Garland (BWG)
syndrome. Isolated left anterior descending (LAD) or circum-
flex (Cx) arteries originating from the pulmonary artery are
even more rare. These anomalies may cause myocardial
ischaemia, myocardial infarction, arrhythmia and sudden
death. Even if the patient is asymptomatic, he/she should
undergo corrective surgery. Here we present the case of an
18-
year-old male who survived sudden cardiac arrest during
exercise. We identified intra-myocardial blood flow from
transthoracic echocardiography, and performed coronary
and computed tomographic (CT) angiography, which showed
that all the coronary arteries were ectatic and curly and there
were disseminated collaterals among the coronary arteries.
We diagnosed ‘anomalous left coronary artery from the
pulmonary artery’ (ALCAPA) syndrome, as additionally, the
LAD originated from the pulmonary artery. We treated the
patient with a left internal mammarian artery – left anterior
descending artery (LIMA–LAD) graft.
Keywords:
coronary artery anomalies, echocardiography,
angiography, surgical treatment
Submitted 18/10/10, accepted 3/5/12
Cardiovasc J Afr
2012;
23
:
e15–e18
DOI: 10.5830/CVJA-2012-037
Bland–White–Garland (BWG) syndrome is a rare congenital
coronary artery anomaly in which the left coronary arterial system
originates from the arteria pulmonalis. Isolated left anterior
descending (LAD) or circumflex (Cx) arteries originating from
the pulmonary artery are even more rare.
1
These anomalies may
cause myocardial ischaemia, myocardial infarction, arrhythmia
and sudden death.
Case report
An 18-year-old male experienced sudden cardiac arrest during
exercise at school. Cardiopulmonary resuscitation was performed
and he was immediately revived. He was hospitalised and at first
cranial pathology was suspected but a cerebral CT scan was
normal. The patient was referred to our clinic with an early
diagnosis of hypertrophic cardiomyopathy.
He did not have any irregularities in his history. His heart rate
was 74 beats per minute, and blood pressure was 110/70 mmHg.
All other examination findings were normal. Electrocardiography
(
ECG) revealed a sinus rhythm and T-wave negativity in the
derivation of V
1
–
V
2
.
His blood analyses were normal. Holter
ECG examination showed a large ORS tachycardia attack at a
rate of five beats/137 per minute.
Transthoracic echocardiography (TTE) revealed a dilated left
ventricle (left ventricular diastolic diameter
=
58
mm, systolic
diameter
=
38
mm) and eccentric left ventricular hypertrophy
with a normal ejection fraction. Colour flow transthoracic
Doppler echocardiography (TTE) demonstrated blood flow in
the myocardial wall, and the parasternal short-axis view showed
abnormal flow towards the pulmonary artery, 1–2 cm below the
pulmonary valve (Fig. 1A, B, C).
Upon suspicion of a coronary artery anomaly, coronary and
CT coronary angiography were performed. The ostium of the
LAD artery was not seen in the coronary angiography. It showed
that the right coronary artery was dilated and tortuous, and it
provided extensive collaterals to the LAD artery, which drained
into the main pulmonary artery. Pulmonary angiography showed
that the left system originated from the pulmonary artery (Fig. 2).
CT coronary angiography confirmed that the LAD artery
originated from the main pulmonary artery (Fig. 3). Cardiac
magnetic resonance imaging (MRI) demonstrated left ventricular
hypertrophy and a sub-endocardial infarct in the apical anterior
and anteroseptal zones.
Surgical correction was planned. The LAD was detached
from the pulmonary artery and connected to the aorta, and the
left internal mammarian artery (LIMA) was anastomosed to the
LAD artery (Fig. 4).
Department of Cardiology, Medical Faculty of Kocaeli
University, Kocaeli, Turkey
TAYFUN SAHİN, MD,
SERDAR BOZYEL, MD
ESER ACAR, MD
ULAS BİLDİRİCİ, MD
ERTAN URAL, MD
DİLEK URAL, MD
Department of Cardiovascular Surgery, Medical Faculty of
Kocaeli University, Kocaeli, Turkey
SADAN YAVUZ, MD
OZGUR BARIS, MD
