一名70岁男性出现源自肺动脉的异常左冠状动脉(ALCAPA):病例报告

Late presentation of an anomalous left coronary artery originating from the pulmonary artery (ALCAPA) in a 70-year-old male: a case report.

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摘要(中文译文)

左冠状动脉异常起源于肺动脉(ALCAPA),又称Bland-White-Garland综合征,是一种罕见的先天性畸形,估计发病率约为每30万活产婴儿中1例。超过90%的未治疗患者在出生后第一年内死亡,存活至成年晚期极为罕见,仅有少数病例报道存活超过六十岁。此类晚期表现具有重要临床意义,因为它们展示了广泛侧支循环在存活中的作用,并带来了独特的诊断和治疗挑战。我们报道一例70岁伊朗男性患者,因持续12小时的急性胸痛就诊。入院时生命体征平稳,心电图显示窦性心律伴多导联ST段压低。连续肌钙蛋白测定均为阴性,经胸超声心动图未见异常。由于持续的临床怀疑,遂行冠状动脉计算机断层扫描血管成像,结果显示左冠状动脉异常起源于肺动脉,右冠状动脉显著扩张并向左侧冠状动脉系统供血形成侧支循环。患者成功接受了左主干冠状动脉再植术至主动脉,出院时病情稳定。本病例为目前报道的年龄最大的ALCAPA诊断之一,强调了即使对表现为非典型急性冠脉综合征的老年患者,也应保持对先天性畸形的高度警惕。本病例为缺乏既往干预而长期存活的有限文献增添了资料,并强调了冠状动脉计算机断层扫描血管成像(CCTA)在无创诊断和手术规划中的关键作用。

Abstract

Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA), also known as Bland-White-Garland syndrome, is a rare congenital anomaly with an estimated incidence of approximately 1 in 300,000 live births. More than 90% of untreated patients die within the first year of life, and survival into late adulthood is exceptionally uncommon, with only a few cases reported beyond the sixth decade. Such late presentations are clinically important, because they demonstrate the role of extensive collateral circulation in survival and pose unique diagnostic and therapeutic challenges. We report the case of a 70-year-old Iranian man who presented with acute chest pain lasting 12 hours. On admission, his vital signs were stable, and electrocardiography revealed sinus rhythm with ST-segment depression in multiple leads. Serial troponin measurements were negative, and transthoracic echocardiography showed no abnormalities. Owing to persistent clinical suspicion, coronary computed tomography angiography was performed, which revealed anomalous origin of the left coronary artery from the pulmonary artery, with a markedly dilated right coronary artery supplying collaterals to the left coronary system. The patient underwent successful surgical reimplantation of the left main coronary artery into the aorta and was discharged in stable condition. This case represents one of the oldest reported diagnoses of ALCAPA, underscoring the importance of maintaining a high index of suspicion for congenital anomalies even in elderly patients with atypical acute coronary presentations. It contributes to the limited literature on late survival without prior intervention and emphasizes the pivotal role of coronary computed tomography angiography (CCTA) in noninvasive diagnosis and surgical planning.

如何引用

AMA

Seyedeh Zeinab Seyedi, Ali Abedi, Bita Mollashahi, Mohammad Namazinia, Seyed Javad Davari-Sani. Late presentation of an anomalous left coronary artery originating from the pulmonary artery (ALCAPA) in a 70-year-old male: a case report.. Journal of medical case reports. 2026; doi:10.1186/s13256-026-06367-3.

APA

Seyedeh Zeinab Seyedi, Ali Abedi, Bita Mollashahi, Mohammad Namazinia, Seyed Javad Davari-Sani (2026). Late presentation of an anomalous left coronary artery originating from the pulmonary artery (ALCAPA) in a 70-year-old male: a case report.. Journal of medical case reports. https://doi.org/10.1186/s13256-026-06367-3

GB/T 7714

Seyedeh Zeinab Seyedi, Ali Abedi, Bita Mollashahi, Mohammad Namazinia, Seyed Javad Davari-Sani. Late presentation of an anomalous left coronary artery originating from the pulmonary artery (ALCAPA) in a 70-year-old male: a case report.[J]. Journal of medical case reports, 2026 doi:10.1186/s13256-026-06367-3.

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