An infantile case of myocardial ischemia associated with hypoplastic left coronary cusp

Q4 Medicine

Journal of Cardiology Cases Pub Date : 2025-05-01 DOI:10.1016/j.jccase.2025.02.001

Hiroyuki Yamada MD , Jun Maeda MD, PhD , Masaru Miura MD, PhD , Yukihiro Yoshimura MD, PhD , Kazuhiko Shibuya MD, PhD

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Abstract

We report a fatal case of myocardial ischemia in a 1-month-old infant caused by a hypoplastic left coronary cusp, a rare, congenital anomaly. The infant presented with acute respiratory failure and hemodynamic instability. Transpericardial echocardiography revealed occlusion of the left coronary ostium by a hypoplastic left cusp. Despite emergency aortic valvuloplasty, refractory cardiogenic shock developed, resulting in death. The present case highlights the importance of considering hypoplastic coronary cusp in the differential diagnosis of infantile myocardial ischemia and the need for careful echocardiographic evaluation of suspected cases.

Learning objective

Hypoplastic aortic valve cusp should be borne in mind as a potential cause of myocardial ischemia in children.

Meticulous echocardiographic assessment should be performed for any coronary artery of anomalous origin and for the morphology of the aortic valve cusp.

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小儿心肌缺血合并左冠状动脉尖发育不全1例

我们报告一个致命的病例心肌缺血在1个月大的婴儿引起的发育不良的左冠状动脉尖，一种罕见的，先天性异常。婴儿表现为急性呼吸衰竭和血流动力学不稳定。经心包超声心动图显示左冠状动脉口被左尖发育不全阻塞。尽管紧急主动脉瓣成形术，难治性心源性休克发生，导致死亡。本病例强调考虑冠状动脉尖发育不全在婴幼儿心肌缺血鉴别诊断中的重要性，以及对疑似病例进行仔细超声心动图评估的必要性。学习目的：儿童主动脉瓣尖发育不全是引起心肌缺血的潜在原因。对于任何异常起源的冠状动脉和主动脉瓣尖的形态，应进行细致的超声心动图评估。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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