A Rare Case of an Adult Pregnant Patient with the Left Coronary Artery Originating from the Pulmonary Artery: Successful Management and Healthy Maternal-Fetal Outcome
Keywords:congenital heart disease, the left coronary artery originates from the pulmonary artery, pregnancy, surgical treatment
An anomalous left coronary artery originating from the pulmonary artery (ALCAPA) refers to the abnormal origin of the left coronary artery either from the main pulmonary artery, pulmonary artery sinus, or the left and right pulmonary arteries, with the main pulmonary artery or pulmonary artery sinus being the most common sites. If not diagnosed and treated promptly, this condition can result in death within the first year of life in 90% of patients. Asymptomatic children can survive into adulthood, but they are at a high risk of sudden death. In this article, we report a case of a 24-year-old pregnant woman who was diagnosed with ALCAPA during prenatal examination. The pregnancy was successfully maintained until 36 weeks, after which a cesarean section was performed. The patient was then admitted to the cardiac surgery department to improve cardiac function, and six weeks later, a successful left coronary artery transplantation was performed. The patient was discharged and followed up for three months, during which her condition remained stable.
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. Journal of the American Society of Echocardiography. 2017; 30: 896–903.
Riley DS, Barber MS, Kienle GS, Aronson JK, von Schoen-Angerer T, Tugwell P, et al. CARE guidelines for case reports:explanation and elaboration document. Journal of Clinical Epi-Demiology. 2017; 89: 218–235.
Bland EF, White PD, Garland J. Congenital anomalies of the coronary arteries: report of an unusual case associated with cardiac hypertrophy. American Heart Journal. 1933; 8: 787–801.
Lin S, Xie M, Lv Q, Wang J, He L, Wang B, et al. Misdiagnosis of anomalous origin of the left coronary artery from the pulmonary artery by echocardiography: Single‐center experience from China. Echocardiography. 2020; 37: 104–113.
Chigurupati K, Gadhinglajkar S, Sreedhar R, Karunakaran J, Dharan BS. Anomalous Origin of the Left Coronary Artery From the Pulmonary Artery in an Infant and Adult: Intraoperative Echocardiographic Comparison. Journal of Cardiothoracic and Vascular Anesthesia. 2016; 30: 1353–1357.
Yuan X, Li B, Sun H, Yang Y, Meng H, Xu L, et al. Surgical outcome in adolescents and adults with anomalous left coronary artery from pulmonary artery. The Annals of Thoracic Surgery. 2018; 106: 1860–1867.
Alexi-Meskishvili V, Berger F, Weng Y, Lange PE, Hetzer R. Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in Adults. Journal of Cardiac Surgery. 1995; 10: 309–315.
Prandi FR, Zaidi AN, LaRocca G, Hadley M, Riasat M, Anastasius MO, et al. Sudden cardiac arrest in an adult with anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA): case report. International Journal of Environmental Research and Public Health. 2022; 19: 1554.
Stout KK, Daniels CJ, Aboulhosn JA, Bozkurt B, Broberg CS, Colman JM, et al. 2018 AHA/ACC Guideline for the Management of Adults With Congenital Heart Disease: A Report of the American College of Cardiology/American Heart Association Task Force on Clinical Practice Guidelines. Circulation. 2019; 139: e698–e800.
Frigault J, Lafrenière-Bessi V, Perron J, Bédard É, Philippon F, Poirier P, et al. Anomalous Left Coronary Artery from the Pulmonary Artery: Masquerading as Peripartum Cardiomyopathy. Annals of Thoracic Surgery. 2018; 106: e33–e35.
Frommelt MA, Miller E, Williamson J, Bergstrom S. Detection of septal coronary collaterals by color flow Doppler mapping is a marker for anomalous origin of a coronary artery from the pulmonary artery. Journal of the American Society of Echocardiography. 2002; 15: 259–263.
Boutsikou M, Shore D, Li W, Rubens M, Pijuan A, Gatzoulis MA, et al. Anomalous left coronary artery from the pulmonary artery (ALCAPA) diagnosed in adulthood: Varied clinical presentation, therapeutic approach and outcome. International Journal of Cardiology. 2018; 261: 49–53.
Ramage K, Grabowska K, Silversides C, Quan H, Metcalfe A. Association of Adult Congenital Heart Disease with Pregnancy, Maternal, and Neonatal Outcomes. JAMA Network Open. 2019; 2: e193667.
Chan A, Wolfe DS, Zaidi AN. Pregnancy and Congenital Heart Disease: A Brief Review of Risk Assessment and Management. Clinical Obstetrics and Gynecology. 2020; 63: 836–851.
Ruys TP, Cornette J, Roos-Hesselink JW. Pregnancy and delivery in cardiac disease. Journal of cardiology. 2013; 61: 107–112.
Yamasaki K, Sawatari H, Konagai N, Kamiya CA, Yoshimatsu J, Muneuchi J, et al. Peripartum Management of Pregnant Women with Congenital Heart Disease. Circulation Journal. 2019; 83: 2257–2264.
Ramírez S, Curi-Curi PJ, Calderón-Colmenero J, García J, Britton C, Erdmenger J, et al. Outcomes of Coronary Reimplantation for Correction of Anomalous Origin of Left Coronary Artery from Pulmonary Artery. Revista EspañOla de Cardiología. 2011; 64: 681–687.
Mavroudis C. Anomalous Origin of the Left Coronary Artery From the Pulmonary Artery Presenting in Adulthood: A French Nationwide Retrospective Study, an Editorial Commentary. Seminars in Thoracic and Cardiovascular Surgery. 2017. (in press)
Qiu J, Li S, Yan J, Wang Q, Song Y, Sun H, et al. Repair of anomalous coronary artery from the pulmonary artery: a-signal center 20-year experience. International Journal of Cardiology. 2016; 223: 625–629.