EARLY DIAGNOSIS AND TREATMENT OF ANOMALOUS ORIGIN OF THE LEFT CORONARY ARTERY
DOI:
https://doi.org/10.46793/PP180321014PKeywords:
Anomalous origin of left coronary artery, ALCAPA, cardiomyopathy, ischemia, infantAbstract
Anomalous origin of the left coronary artery from pulmonary artery (ALCAPA) presents very rare congenital anomaly, with prevalence rate of 1:300.000 live births, and in such a case myocardial ischemia occurs due to phenomenon called » steal phenomenon«.
Two months old infant was admitted with signs of serious congestive heart insufficiency, followed by metabolic acidosis, highly elevated cardiospecific enzymes and proteins, heart enlargement on chest X ray and electrocardiogram revealed miocardial ischemia. Echocardiography showed significant dilatation and hypocontractility of left ventricle caused by anomalous origin of left conorary artery from pulmonary artery in presence of phenomenon « steal phenomenon». Diagnosis was confirmed by conorary angiography. At age three and half months cardiosurgical intervention was performed – transposition of left coronary artery. Child had normal postoperative recovering and excellent long duration outcome. Check up visits during two years of follow up showed complet recovry.
In nontreated patients with ALCAPA mortality rate in a first year of life is 90%. However, prognosis has been significantly improved as a result of early echocardiographic diagnosis and accomplished surgical intervention.
References
Brooks H. Two cases of an abnormal coronary artery of the heart arising from the pulmonary artery: with some remarks upon the effects of this anomaly in producing cirsoid dilation of the vessel. J Anat 1882;20:26-9.
Bland EF, White PD, Garland J. Congenital anomalies of the coronary arteries: report of an unusual case associated with cardiac hypertrophy. 1933;8:787-801.
Cabrera A, Chen D, Pignatelli R, et al. Outcomes of anomalous left coronary artery from pulmonary artery repair: beyond normal function. Ann Thorac Surg 2015; 99:1342-7
Bjelakovic B, Pejcic L, Parezanovic V. Vomiting as the initial clinical presentation of myocardial infarction in children with anomalous left coronary artery from the pulmonary trunk. Turk J Pediatr 2008; 50(5):476-9.
5. Patel SG, Frommelt MA, Frommelt PC, Kutty S, Cramer JW. Echocardiographic Diagnosis, Surgical Treatment, and Outcomes of Anomalous Left Coronary Artery from the Pulmonary Artery. J Am Soc Echocardiogr 2017; 30:896-903.
Weigand J, Marshall CD, Bacha EA, Chen JM, Richmond ME. Repair of Anomalous Left Coronary Artery From the Pulmonary Artery in the Modern Era: Preoperative Predictors of Immediate Postoperative Outcomes and Long Term Cardiac Follow-up. Pediatric Cardiology 2015; 36:489–9.
Lange R, Vogt M, Hörer J, Cleuziou J, Menzel A, Holper K, Hess J and Schreiber C. Long-Term Results of Repair of Anomalous Origin of the Left Coronary Artery From the Pulmonary Artery. Ann Thorac Surg 2007; 83:1463-71
Lange R, Cleuziou J, Krane M, Ewert P, Pabst von Ohain J, Beran E, et al. Long-term outcome after anomalous left coronary artery from the pulmonary artery repair: a 40-year single-centre experience. Eur J Cardiothorac Surg 2018; 53:732-9.
Nordmeyer S, Schmitt B, Nasseri B, Alexi-Meskishvili V, Kuehne T, Berger F, Nordmeyer J. Presence of reduced regional left ventricular function even in the absence of left ventricular wall scar tissue in the long term after repair of an anomalous left coronary artery from the pulmonary artery. Cardiol Young 2018; 28:200-7.
Fratz S, Hager A, Schreiber C, Schwaiger M, Hess J, Stern HC. Long-term myocardial scarring after operation for anomalous left coronary artery from the pulmonary artery. Ann Thorac Surg 2011; 92:1761-5.
