TY - JOUR
T1 - Anomalous coronary artery from the pulmonary artery diagnosed in adulthood
T2 - a case series on variations of coronary anatomy and the diagnostic value of cardiac magnetic resonance imaging
AU - Talebian Yazdi, Mehrdad
AU - Robbers-Visser, D.
AU - van der Bilt, I. A. C.
AU - Boekholdt, S. M.
AU - Koolbergen, D. R.
AU - Planken, R. N.
AU - Groenink, M.
N1 - © The Author(s) 2022. Published by Oxford University Press on behalf of the European Society of Cardiology.
PY - 2022/8/1
Y1 - 2022/8/1
N2 - Background Anomalous coronary artery from the pulmonary artery is a rare congenital disorder with high mortality rates in infancy. Adult cases can present with life-threatening arrhythmias and sudden cardiac death. Case summary We report three cases of adults with anomalous pulmonary origin of each of the main coronary branches. The first patient with an anomalous left coronary artery from the pulmonary artery presented with an out-of-hospital cardiac arrest. The second patient with an anomalous circumflex artery from the pulmonary artery was evaluated for a bicuspid aortic valve and a suspected coronary fistula but was otherwise asymptomatic. The third patient with an anomalous right coronary artery from the pulmonary artery presented with anginal symptoms. In all cases, the diagnosis was made by cardiac computed tomography or coronary angiography. Cardiac magnetic resonance imaging was performed in all patients to guide clinical decision making on surgical or non-invasive management. All patients underwent surgical repair. In two patients, a dual coronary artery system was restored by aortic reimplantation of the anomalous coronary artery. In one patient, aortic reimplantation was unsuccessful due to poor vessel quality and the anomalous coronary artery was ligated. Clinical follow-up during 1.8–9.7 years did not show any cardiovascular complications and all patients are currently alive and asymptomatic. Discussion Anomalous coronary artery from the pulmonary artery can have various clinical presentations in adulthood. Cardiac magnetic resonance imaging is a useful modality to guide selection of patients who might have symptomatic or prognostic benefit from surgical repair.
AB - Background Anomalous coronary artery from the pulmonary artery is a rare congenital disorder with high mortality rates in infancy. Adult cases can present with life-threatening arrhythmias and sudden cardiac death. Case summary We report three cases of adults with anomalous pulmonary origin of each of the main coronary branches. The first patient with an anomalous left coronary artery from the pulmonary artery presented with an out-of-hospital cardiac arrest. The second patient with an anomalous circumflex artery from the pulmonary artery was evaluated for a bicuspid aortic valve and a suspected coronary fistula but was otherwise asymptomatic. The third patient with an anomalous right coronary artery from the pulmonary artery presented with anginal symptoms. In all cases, the diagnosis was made by cardiac computed tomography or coronary angiography. Cardiac magnetic resonance imaging was performed in all patients to guide clinical decision making on surgical or non-invasive management. All patients underwent surgical repair. In two patients, a dual coronary artery system was restored by aortic reimplantation of the anomalous coronary artery. In one patient, aortic reimplantation was unsuccessful due to poor vessel quality and the anomalous coronary artery was ligated. Clinical follow-up during 1.8–9.7 years did not show any cardiovascular complications and all patients are currently alive and asymptomatic. Discussion Anomalous coronary artery from the pulmonary artery can have various clinical presentations in adulthood. Cardiac magnetic resonance imaging is a useful modality to guide selection of patients who might have symptomatic or prognostic benefit from surgical repair.
UR - https://www.scopus.com/inward/record.uri?partnerID=HzOxMe3b&scp=85141741058&origin=inward
UR - https://www.ncbi.nlm.nih.gov/pubmed/36045648
U2 - https://doi.org/10.1093/ehjcr/ytac345
DO - https://doi.org/10.1093/ehjcr/ytac345
M3 - Article
C2 - 36045648
SN - 2514-2119
VL - 6
SP - ytac345
JO - European Heart Journal - Case Reports
JF - European Heart Journal - Case Reports
IS - 8
M1 - oeac065
ER -