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      Different Presentation of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in Adults: Case Reports

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          Abstract

          Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital cardiac malformation. We report three cases of ALCAPA who survived to adulthood. The first case was a 51-year-old woman who complained of typical chest pain that was diagnosed with ALCAPA using cardiac catheterization and coronary computed tomographic angiography (CTA). The second case was a 30-year-old woman with a history of surgery for atrial septal defect at 10 years old who presented with progressive exertional dyspnea. Cardiac catheterization confirmed the diagnosis of ALCAPA. The third case was a 19-year-old man who was brought to our clinic due to aborted sudden cardiac death on the previous day. Cardiac catheterization and coronary CTA confirmed the diagnosis. They underwent the closure of orifice of the anomalous left coronary artery and grafting the left anterior descending artery concomitantly with mitral valve repair. All patients were followed up during a mean of 8.7 months and they were asymptomatic.

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          Anomalous origin of the left coronary artery from the pulmonary artery: collective review of surgical therapy.

          Anomalous origin of the left coronary artery from the pulmonary artery is an extremely rare but potentially fatal congenital coronary anomaly. Prompt surgical reestablishment of a two-coronary system on diagnosis yields excellent results and allows progressive and nearly total myocardial recovery. Follow-up of all patients is required to assess the adequacy of repair and to exclude ongoing or recurrent myocardial insult.
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            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.

            Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital coronary abnormality associated with early infant mortality and adult sudden death. As it predominantly presents in the first year of life, diagnosis in living adults is extremely rare. Current management is based on limited case series or extrapolated from pediatric cases. Modern advances in noninvasive cardiac imaging have substantially increased the number of diagnoses, uncovering a large adult population that has not been reviewed. The availability of newer diagnostic modalities correlates with an increasing incidence in an older cohort, and true association between sudden death and ALCAPA may be lower, especially among older patients. A comprehensive literature search was performed for all case reports of ALCAPA on MEDLINE and PubMed using the keywords ALCAPA, Bland-White-Garland, and coronary anomaly; and augmented by references from published case reports from 1908 to 2008. All adult cases, defined by age 18 years and older, were reviewed for this article. One hundred fifty-one adult cases of ALCAPA are described, in addition to the case of an asymptomatic 53-year-old woman. The average reported age was 41 years old with the oldest being 83. Sixty-six percent of the patients presented with symptoms of angina, dyspnea, palpitations, or fatigue; 17% presented with ventricular arrhythmia, syncope, or sudden death; and 14% were asymptomatic. Twelve percent were diagnosed at autopsy. The majority had some form of surgical correction during their clinical course. ALCAPA is a rare and life-threatening condition in adults. The availability of newer, less invasive diagnostic modalities has resulted in more frequent identification of this condition in an older cohort. © 2011 Wiley Periodicals, Inc.
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              Anomalous origin of the left coronary artery from the pulmonary artery (Bland-White-Garland syndrome) in adult patients: long-term follow-up after surgery.

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                Author and article information

                Journal
                Iran J Med Sci
                Iran J Med Sci
                Iranian Journal of Medical Sciences
                Iranian Journal of Medical Sciences (Iran )
                0253-0716
                1735-3688
                November 2017
                : 42
                : 6
                : 599-602
                Affiliations
                [1 ]Department of Cardiovascular Surgery, Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
                [2 ]Heart Valve Disease Research Center, Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
                [3 ]Echocardiography Research Center, Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
                [4 ]Department of Cardiac Anesthesia, Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
                [5 ]Department of Radiology, Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
                Author notes
                Correspondence: Alireza Alizadeh Ghavidel, MD; Heart Valve Disease Research Center, Rajaie Cardiovascular Medical and Research Center, Vali-e-Asr Avenue, Tehran 19969-11151, Iran Tel: +98 912 1590518 Fax: +98 21 22663209 Email: aaghavidel@ 123456gmail.com
                Article
                IJMS-42-599
                5684382
                bb1fbb92-e4f0-49cc-8d1d-45efd30b826e
                Copyright: © Iranian Journal of Medical Sciences

                This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 07 August 2016
                : 21 August 2016
                : 14 June 2016
                Categories
                Case Report

                Medicine
                coronary vessel anomalies,heart defects,congenital
                Medicine
                coronary vessel anomalies, heart defects, congenital

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