Talebian Yazdi Mehrdad, Robbers-Visser D, van der Bilt I A C, Boekholdt S M, Koolbergen D R, Planken R N, Groenink M
Department of Cardiology, Alrijne Hospital, Simon Smitweg 1, 2353 GA Leiderdorp, The Netherlands.
Department of Clinical and Experimental Cardiology, Amsterdam UMC, University of Amsterdam, Meibergdreef 9, 1105 BK Amsterdam, The Netherlands.
Eur Heart J Case Rep. 2022 Aug 16;6(8):ytac345. doi: 10.1093/ehjcr/ytac345. eCollection 2022 Aug.
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.
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.
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.
起源于肺动脉的异常冠状动脉是一种罕见的先天性疾病，婴儿期死亡率很高。成人病例可出现危及生命的心律失常和心源性猝死。
我们报告3例主要冠状动脉分支均起源于肺动脉的成人病例。第一例左冠状动脉起源于肺动脉的患者出现院外心脏骤停。第二例回旋支起源于肺动脉的患者因二叶式主动脉瓣和疑似冠状动脉瘘接受评估，但无其他症状。第三例右冠状动脉起源于肺动脉的患者出现心绞痛症状。所有病例均通过心脏计算机断层扫描或冠状动脉造影确诊。所有患者均接受了心脏磁共振成像检查，以指导关于手术或非侵入性治疗的临床决策。所有患者均接受了手术修复。2例患者通过将异常冠状动脉重新植入主动脉恢复了双冠状动脉系统。1例患者因血管质量差，主动脉重新植入术未成功，异常冠状动脉被结扎。1.8至9.7年的临床随访未显示任何心血管并发症，所有患者目前均存活且无症状。
起源于肺动脉的异常冠状动脉在成年期可有多种临床表现。心脏磁共振成像是一种有用的检查方法，可指导选择可能从手术修复中获得症状改善或预后益处的患者。
