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A rare case of anomalous left circumflex coronary artery from the left pulmonary artery
International Journal of Cardiology 88 (2003) 305–307 www.elsevier.com / locate / ijcard
Letter to the Editor
A rare case of anomalous left circumflex coronary artery from the left pulmonary artery a, a a a b c Jinyoung Song *, Jaeyoung Lee , Soojin Kim , Woosup Shim , Woonghwan Kim , Yangmin Kim a
b
Pediatric Cardiology, Sejong Hospital, Sejong Heart Institution, Sejong, South Korea Cardiovascular Surgery, Sejong Hospital, Sejong Heart Institution, Sejong, South Korea c Cardiac Radiology, Sejong Hospital, Sejong Heart Institution, Sejong, South Korea Received 2 February 2002; accepted 6 April 2002
Abstract The anomalous origin of the coronary artery in children is a rare disease. In particular, an anomalous origin of the left coronary artery from the pulmonary artery is a fatal condition in neonate with myocardial failure. A 46-month-old child with an anomalous origin of the left circumflex artery from the left pulmonary artery was detected due to asymptomatic cardiac murmur and immediately re-implantation of the left circumflex coronary artery was carried out successfully. 2002 Elsevier Science Ireland Ltd. All rights reserved.
1. Case report A symptom-free girl (3 years and 10 months of age) was referred because of cardiac murmur which had been detected incidentally. She looked healthy and weighed 14 kg although ligation of patent ductus arteriosus was carried out when she was 13 months old. When she was presented, her vital signs were normal and normal findings of physical examination were detected except grade 2 systolic murmur on the left lower sternal border. Her electrocardiogram showed only incomplete right bundle branch block (Fig. 1) and a normal chest radiograph was present. A two-dimensional echocardiogram detected abnormal retrograded color flow to the left pulmonary artery with slightly dilated left *Corresponding author. Present address: Kyunggido bucheonsi sosagu sosa 2 dong 91-121, 422-711 South Korea. Tel.: 182-32-340-1125; fax: 182-32-340-1236. E-mail address: [email protected] (J. Song).
coronary artery and a minimal degree of aortic regurgitation. But internal diameters of the left ventricle were within normal limits with 63% of left ventricle ejection fraction. The clinical diagnosis was that of a small coronary arteriovenous fistula and pressure measured at the cardiac catheterization revealed elevated left ventricular end-diastolic pressure with mild pulmonary hypertension (left ventricle 106 / 22 mmHg and left pulmonary artery 66 / 33 mmHg). There was a 5% (70→75%) increase in oxygen saturation in the left pulmonary artery. Angiography of the aorta showed that the left circumflex coronary artery appeared to fill slightly later than the left descending and right coronary arteries via collaterals from the left anterior descending coronary artery (Fig. 2). Selective coronary angiography showed an abnormal origin of the left circumflex artery from the left pulmonary artery. But the left ventriculogram showed that of normal contraction. A Dipyridamole stress thallium scan showed decreased
0167-5273 / 02 / $ – see front matter 2002 Elsevier Science Ireland Ltd. All rights reserved. doi:10.1016/S0167-5273(02)00390-X
306
J. Song et al. / International Journal of Cardiology 88 (2003) 305–307
Fig. 1. The electrocardiogram of the present case showed incomplete right bundle branch block without any definite evidence of myocardial ischemia.
uptake in the basal lateral wall and inferolateral wall of the left ventricle with reversibility. After surgical re-implantation was recommended, her abnormal left circumflex coronary artery was successfully re-implanted into the aorta. Postoperatively, she was in good health and discharged uneventfully.
2. Discussion The anomalous origin of the left circumflex coronary artery from the pulmonary artery is such a rare congenital coronary anomaly that only 11 cases have been reported [1,2]. The cardiac symptoms which the patients complained of and abnormalities in their electrocardiograms could be attributed to coronary anomaly. In our case, the correct diagnosis was delayed until angiography of the aortic root was carried out because we found the origins of both coronary arteries in 2-D and color Doppler echocardiogram that made us diagnose our case as a coronary arteriovenous fistula. Although a resting echocardiogram of the present case revealed normal
Fig. 2. The anterior–posterior (top) and lateral (bottom) angiograms of the aortic root revealed a left circumflex coronary artery from the left pulmonary artery with multiple collaterals (filled arrows).
motion of the left ventricle, increased left ventricular end-diastolic pressure in cardiac catheterization and reversible abnormality in stress thallium scan showed myocardial ischemic damage as we could have
J. Song et al. / International Journal of Cardiology 88 (2003) 305–307
expected. Her patent ductus arteriosus could be helpful not only for myocardial perfusion until ligation and also for the development of the collaterals which could result in the coronary steal phenomenon [3]. We found only one patient with anomalous origin of the circumflex artery from the left pulmonary artery like the present case among the total 11 cases. In these coronary anomalies, it is thought that surgical treatment should be performed as soon as possible not only due to myocardial ischemia but also due to malignant ventricular arrhythmia which could lead to sudden cardiac death in adult life [4]. Because establishment of dual coronary circulation is better than simple ligation [5], we re-implanted the anomalous coronary artery to the aorta successfully. And we can hopefully expect complete recovery from myocardial dysfunction.
307
References [1] Ott DA, Cooley DA, Pinsky WW, Mullins CE. Anomalous origin of circumflex coronary artery from the right pulmonary artery: report of a rare anomaly. J Thorac Cardiovasc Surg 1978;76:190–4. [2] Alexi-Meskishvili V, Dahnert I, Hetzer R, Lange PE, Karl TM. Origin of the circumflex coronary artery from the pulmonary artery in infants. Ann Thorac Surg 1998;66:1406–9. [3] Garcia CM, Chandler J, Russel R. Anomalous left coronary artery from the right pulmonary artery: First adult case report. Am Heart J 1992;123:526–8. [4] Frapier JM, Leclercq F, Bodino M, Chaptal PA. Malignant ventricular arrhythmias revealing anomalous origin of the left coronary artery from the pulmonary artery in two adults. Eur J Cardiothorac Surg 1999;15:539–41. [5] Schwartz ML, Jonas RA, Colan SD. Anomalous origin of left coronary artery from pulmonary artery: Recovery of left ventricular function after dual coronary repair. J Am Coll Cardiol 1997;30:547– 53.
Letter to the Editor
A rare case of anomalous left circumflex coronary artery from the left pulmonary artery a, a a a b c Jinyoung Song *, Jaeyoung Lee , Soojin Kim , Woosup Shim , Woonghwan Kim , Yangmin Kim a
b
Pediatric Cardiology, Sejong Hospital, Sejong Heart Institution, Sejong, South Korea Cardiovascular Surgery, Sejong Hospital, Sejong Heart Institution, Sejong, South Korea c Cardiac Radiology, Sejong Hospital, Sejong Heart Institution, Sejong, South Korea Received 2 February 2002; accepted 6 April 2002
Abstract The anomalous origin of the coronary artery in children is a rare disease. In particular, an anomalous origin of the left coronary artery from the pulmonary artery is a fatal condition in neonate with myocardial failure. A 46-month-old child with an anomalous origin of the left circumflex artery from the left pulmonary artery was detected due to asymptomatic cardiac murmur and immediately re-implantation of the left circumflex coronary artery was carried out successfully. 2002 Elsevier Science Ireland Ltd. All rights reserved.
1. Case report A symptom-free girl (3 years and 10 months of age) was referred because of cardiac murmur which had been detected incidentally. She looked healthy and weighed 14 kg although ligation of patent ductus arteriosus was carried out when she was 13 months old. When she was presented, her vital signs were normal and normal findings of physical examination were detected except grade 2 systolic murmur on the left lower sternal border. Her electrocardiogram showed only incomplete right bundle branch block (Fig. 1) and a normal chest radiograph was present. A two-dimensional echocardiogram detected abnormal retrograded color flow to the left pulmonary artery with slightly dilated left *Corresponding author. Present address: Kyunggido bucheonsi sosagu sosa 2 dong 91-121, 422-711 South Korea. Tel.: 182-32-340-1125; fax: 182-32-340-1236. E-mail address: [email protected] (J. Song).
coronary artery and a minimal degree of aortic regurgitation. But internal diameters of the left ventricle were within normal limits with 63% of left ventricle ejection fraction. The clinical diagnosis was that of a small coronary arteriovenous fistula and pressure measured at the cardiac catheterization revealed elevated left ventricular end-diastolic pressure with mild pulmonary hypertension (left ventricle 106 / 22 mmHg and left pulmonary artery 66 / 33 mmHg). There was a 5% (70→75%) increase in oxygen saturation in the left pulmonary artery. Angiography of the aorta showed that the left circumflex coronary artery appeared to fill slightly later than the left descending and right coronary arteries via collaterals from the left anterior descending coronary artery (Fig. 2). Selective coronary angiography showed an abnormal origin of the left circumflex artery from the left pulmonary artery. But the left ventriculogram showed that of normal contraction. A Dipyridamole stress thallium scan showed decreased
0167-5273 / 02 / $ – see front matter 2002 Elsevier Science Ireland Ltd. All rights reserved. doi:10.1016/S0167-5273(02)00390-X
306
J. Song et al. / International Journal of Cardiology 88 (2003) 305–307
Fig. 1. The electrocardiogram of the present case showed incomplete right bundle branch block without any definite evidence of myocardial ischemia.
uptake in the basal lateral wall and inferolateral wall of the left ventricle with reversibility. After surgical re-implantation was recommended, her abnormal left circumflex coronary artery was successfully re-implanted into the aorta. Postoperatively, she was in good health and discharged uneventfully.
2. Discussion The anomalous origin of the left circumflex coronary artery from the pulmonary artery is such a rare congenital coronary anomaly that only 11 cases have been reported [1,2]. The cardiac symptoms which the patients complained of and abnormalities in their electrocardiograms could be attributed to coronary anomaly. In our case, the correct diagnosis was delayed until angiography of the aortic root was carried out because we found the origins of both coronary arteries in 2-D and color Doppler echocardiogram that made us diagnose our case as a coronary arteriovenous fistula. Although a resting echocardiogram of the present case revealed normal
Fig. 2. The anterior–posterior (top) and lateral (bottom) angiograms of the aortic root revealed a left circumflex coronary artery from the left pulmonary artery with multiple collaterals (filled arrows).
motion of the left ventricle, increased left ventricular end-diastolic pressure in cardiac catheterization and reversible abnormality in stress thallium scan showed myocardial ischemic damage as we could have
J. Song et al. / International Journal of Cardiology 88 (2003) 305–307
expected. Her patent ductus arteriosus could be helpful not only for myocardial perfusion until ligation and also for the development of the collaterals which could result in the coronary steal phenomenon [3]. We found only one patient with anomalous origin of the circumflex artery from the left pulmonary artery like the present case among the total 11 cases. In these coronary anomalies, it is thought that surgical treatment should be performed as soon as possible not only due to myocardial ischemia but also due to malignant ventricular arrhythmia which could lead to sudden cardiac death in adult life [4]. Because establishment of dual coronary circulation is better than simple ligation [5], we re-implanted the anomalous coronary artery to the aorta successfully. And we can hopefully expect complete recovery from myocardial dysfunction.
307
References [1] Ott DA, Cooley DA, Pinsky WW, Mullins CE. Anomalous origin of circumflex coronary artery from the right pulmonary artery: report of a rare anomaly. J Thorac Cardiovasc Surg 1978;76:190–4. [2] Alexi-Meskishvili V, Dahnert I, Hetzer R, Lange PE, Karl TM. Origin of the circumflex coronary artery from the pulmonary artery in infants. Ann Thorac Surg 1998;66:1406–9. [3] Garcia CM, Chandler J, Russel R. Anomalous left coronary artery from the right pulmonary artery: First adult case report. Am Heart J 1992;123:526–8. [4] Frapier JM, Leclercq F, Bodino M, Chaptal PA. Malignant ventricular arrhythmias revealing anomalous origin of the left coronary artery from the pulmonary artery in two adults. Eur J Cardiothorac Surg 1999;15:539–41. [5] Schwartz ML, Jonas RA, Colan SD. Anomalous origin of left coronary artery from pulmonary artery: Recovery of left ventricular function after dual coronary repair. J Am Coll Cardiol 1997;30:547– 53.