Diagnosis and Surgical Outcomes of Patients with Anomalous Left Coronary Artery from the Pulmonary Artery: A Single Taiwanese Medical Center Experience
Background: Anomalous left coronary artery from the pulmonary artery (ALCAPA), a very rare congenital cardiac anomaly, is associated with a high mortality rate among infants who are not diagnosed or treated in a timely manner. Surgical intervention with the reconstruction for a two-coronary-system circulation is the main treatment; however, there have been very few reported cases from Taiwan. In this study, we aim to describe the clinical manifestations, diagnostic methods, surgery types, and surgical outcomes in patients with ALCAPA from a single Taiwanese medical center.
Methods: We retrospectively reviewed patients diagnosed with ALCAPA who underwent surgery at our institution between January 2001 and October 2018. Clinical presentations, noninvasive and invasive study results, surgical methods, and postoperative follow-up results were assessed from medical records. Moreover, literature on this particular cardiovascular anomaly was reviewed.
Results: The study included 6 patients (5 children and 1 adult). The diagnosis was confirmed using cardiac catheterization and coronary angiography in four patients and only echocardiography in two patients. All patients underwent surgical correction and survived. Four patients showed improvements in left ventricular function and mitral regurgitation (MR).
Conclusion: Early diagnosis and timely surgical intervention could avoid mortality regardless of the method of operation. ALCAPA can be definitively diagnosed using noninvasive echocardiography. Both left ventricular systolic function and mitral insufficiency could improve after the surgical intervention in pediatric patients. Repair or replacement of the mitral valve could be reserved for persistent MR complicated with congestive heart failure, particularly in patients who received the initial operation beyond infancy.
Alexi-Meskishvili V, Nasseri BA, Nordmeyer S, et al. 2011. Repair of anomalous origin of the left coronary artery from the pulmonary artery in infants and children. J Thorac Cardiovasc Surg 142:868-74.
Azakie A, Russell JL, McCrindle BW, et al. 2003. Anatomic repair of anomalous left coronary artery from the pulmonary artery by aortic reimplantation: Early survival, patterns of ventricular recovery and late outcome. Ann Thorac Surg 75:1535-41.
Ben Ali W, Metton O, Roubertie F, et al. 2009. Anomalous origin of the left coronary artery from the pulmonary artery: late results with special attention to the mitral valve. Eur J Cardiothorac Surg 36:244-9.
Bland EF, White PD, Garland J. 1933. Congenital anomalies of the coronary arteries: Report of an unusual case associated with cardiac hypertrophy. Am Heart J 8:787-801.
Brooks HSJ. 1885. Two cases of an abnormal coronary artery of the heart, arising from the pulmonary artery: with some remarks upon the effect of this anomaly in producing cirsoid dilatation of the vessels. Trans Acad Med Irel 3:447-9.
Brotherton H, Philip RK. 2008. Anomalous left coronary artery from pulmonary artery (ALCAPA) in infants: A 5-year review in a defined birth cohort. Eur J Pediatr 167:43-6.
Formica F, Amigoni P, Mariani S, et al. 2014. A rare case of ALCAPA and rheumatic mitral valve regurgitation in an adult patient. Heart Surg Forum.17:E250-52.
Gao Y, Zhang J, Huang GY, et al. 2017. Surgical outcomes of anomalous origin of the left coronary artery from the pulmonary artery in children : An echocardiography follow-up. Chin Med J (Engl) 130:2333-8.
Harold JG, Bass TA, Bashore TM, et al. 2013. ACCF/AHA/SCAI 2013 update of the clinical competence statement on coronary artery interventional procedures: a report of the American College of Cardiology Foundation/American Heart Association/American College of Physicians Task Force on Clinical Compete. J Am Coll Cardiol 62:357-96.
Hsu HT and Wu MT. 2014. Bland–White–Garland syndrome. QJM 108:71–2.
Huang CH, Lin YC, Tsai CS, et al. 2017. Aborted sudden cardiac death in a young male with anomalous left coronary artery arising from the pulmonary artery. J Med Sci 37:256-8.
Isomatsu Y, Imai Y, Shin’oka T, et al. 2001. Surgical intervention for anomalous origin of the left coronary artery from the pulmonary artery: the Tokyo experience. J Thorac Cardiovasc Surg121:792-7.
Jiang GP, Wang HF, Gong FQ, et al. 2014. Diagnostic value of parasternal pulmonary artery short-axis view for the anomalous origin of the left coronary artery from the pulmonary artery. J Cardiol 63:444-8.
Jin YD, Hsiung MC, Tsai SK, et al. 2011. Successful intraoperative identification of an anomalous origin of the left coronary artery from the pulmonary artery using real time three-dimensional transesophageal echocardiography. Echocardiography 28:149-51.
Lardhi AA. 2010. Anomalous origin of left coronary artery from pulmonary artery: A rare cause of myocardial infarction in children. J Fam Community Med 17:113-6.
Muzaffar T, Ganie FA, Swamy SG, et al. 2014. The surgical outcome of anomalous origin of the left coronary artery from the pulmonary artery. Int Cardiovasc Res J 8:57-60.
Ojala T, Salminen J, Happonen JM, et al. 2010. Excellent functional result in children after correction of anomalous origin of left coronary artery from the pulmonary artery--a population-based complete follow-up study. Interact Cardiovasc Thorac Surg 10:70-5.
Pachon R, Bravo C, Niemiera M. 2014. Anomalous origin of the left coronary artery from pulmonary artery (ALCAPA). Clin Exp Cardiol 5:10-2.
Peña E, Nguyen ET, Merchant N, et al. 2009. ALCAPA syndrome: Not just a pediatric disease. RadioGraphics 29:553-65.
Rajbanshi BG, Burkhart HM, Schaff HV, et al. 2014. Surgical strategies for anomalous origin of coronary artery from pulmonary artery in adults. J Thorac Cardiovasc Surg148:220-4.
Ramírez S, Curi-Curi PJ, Calderón-Colmenero J, et al. 2011. Outcomes of coronary reimplantation for correction of anomalous origin of left coronary artery from pulmonary artery. Rev Esp Cardiol 64:681-7.
Rodriguez-Gonzalez M, Tirado AM, Hosseinpour R, et al. 2015. Anomalous origin of the left coronary artery from the pulmonary artery: Diagnoses and surgical results in 12 pediatric patients. Texas Hear Inst J 42:350-6.
Su CS, Tsai IC, Lin WW, et al. 2010. Usefulness of multidetector-row computed tomography in diagnosis of anomalous origin of left coronary artery arising from the pulmonary artery. J Chinese Med Assoc 73:492-5.
Talwar S, Jha AJ, Choudhary SK, et al. 2013. Repair of anomalous left coronary artery from pulmonary artery (ALCAPA) beyond infancy. Heart Surg Forum 16:214-20.
Tseng KFY. 2013. Anomalous origin of left coronary artery from the pulmonary trunk in a mildly symptomatic adult female. Case Rep Surg 2013:1-4.
Vossler JD, Entrikin DW, Quartermain MD, et al. 2012. Anomalous left coronary artery from the pulmonary arteryin an adult. Heart Surg Forum 15:284.
Yang H, Li J, Ji X. 2018. Diagnosis of anomalous origin of the left coronary artery from the pulmonary artery with echocardiography and digital subtraction angiography. Case Reports Cardiol 2018:1-6.
Yau JM, Singh R, Halpern EJ, et al. 2011. Anomalous origin of the left coronary artery from the pulmonary artery in adults: A comprehensive review of 151 adult cases and a new diagnosis in a 53-year-old woman. Clin Cardiol 34:204-10.
Zheng JY, Han L, Ding WH, et al. 2010. Clinical features and long-term prognosis of patients with anomalous origin of the left coronary artery from the pulmonary artery. Chin Med J (Engl) 123:2888-94.
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