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An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report

BACKGROUND: An anomalous aortic origin of a coronary artery (AAOCA) has been considered as a dominant cause of sudden cardiac death (SCD) among young age children. Therefore, it is critical to identify AAOCA timely to avoid lethal events. Recently, accumulating cases of right or left coronary arteri...

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Autores principales: Hu, Fan, Wang, Xinyue, Wan, Jiaojiao, Li, Yifei, Wang, Tao, Zhou, Kaiyu, Shi, Xiaoqing, Liu, Zhongqiang, Fang, Jie, Hua, Yimin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9222707/
https://www.ncbi.nlm.nih.gov/pubmed/35757337
http://dx.doi.org/10.3389/fcvm.2022.918832
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author Hu, Fan
Wang, Xinyue
Wan, Jiaojiao
Li, Yifei
Wang, Tao
Zhou, Kaiyu
Shi, Xiaoqing
Liu, Zhongqiang
Fang, Jie
Hua, Yimin
author_facet Hu, Fan
Wang, Xinyue
Wan, Jiaojiao
Li, Yifei
Wang, Tao
Zhou, Kaiyu
Shi, Xiaoqing
Liu, Zhongqiang
Fang, Jie
Hua, Yimin
author_sort Hu, Fan
collection PubMed
description BACKGROUND: An anomalous aortic origin of a coronary artery (AAOCA) has been considered as a dominant cause of sudden cardiac death (SCD) among young age children. Therefore, it is critical to identify AAOCA timely to avoid lethal events. Recently, accumulating cases of right or left coronary arteries originating from inappropriate locations at the sinus of Valsalva have been identified. Here, we report a rare case of AAOCA with an intra-arterial wall course pretending normal migration on imaging screening in a patient who suffered from syncope. CASE SUMMARY: A 7-year-old male without a previous history of cardiovascular and cerebrovascular diseases suddenly suffered from sharp chest pain and syncope after intensive exercise. The electrocardiogram showed that the ST segment of multiple leads was depressed by more than 0.05 mV, and biomarkers indicated severe myocardial injuries. The left ventricular ejection fraction (LVEF) decreased dramatically to 23%. Fulminant myocarditis and cardiomyopathy were therefore excluded. However, a relatively normal coronary artery origin, which arose from the left coronary sinus, presented on echocardiography and cardiac CT angiography (CTA). It is difficult to draw an association between severe clinical manifestations and slight malformations on echocardiography and CTA. Furthermore, selective coronary angiography revealed that an anomalous left coronary artery arose from the superior margin of the inappropriate sinus, developed an intramural wall course and finally exits the left sinus of Valsalva and migrated between the aorta and the pulmonary artery, which induced severe myocardial infarction during exercise. Then, the patient received surgical correction with a modified unroofing procedure. After 2 months of intensive treatment, the patient was discharged and remained asymptomatic through 18 months of follow-up. CONCLUSION: AAOCA, especially anomalous left coronary artery (ALCA), represents a major potential risk of SCD. We reported an atypical manifestation of ALCA arising from the inappropriate sinus of Valsalva and merging into the intra-arterial wall to develop a strange course and then sprout between the aorta and the pulmonary artery. The diversity of AAOCA might present as a relatively normal course under non-invasive radiological imaging scanning.
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spelling pubmed-92227072022-06-24 An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report Hu, Fan Wang, Xinyue Wan, Jiaojiao Li, Yifei Wang, Tao Zhou, Kaiyu Shi, Xiaoqing Liu, Zhongqiang Fang, Jie Hua, Yimin Front Cardiovasc Med Cardiovascular Medicine BACKGROUND: An anomalous aortic origin of a coronary artery (AAOCA) has been considered as a dominant cause of sudden cardiac death (SCD) among young age children. Therefore, it is critical to identify AAOCA timely to avoid lethal events. Recently, accumulating cases of right or left coronary arteries originating from inappropriate locations at the sinus of Valsalva have been identified. Here, we report a rare case of AAOCA with an intra-arterial wall course pretending normal migration on imaging screening in a patient who suffered from syncope. CASE SUMMARY: A 7-year-old male without a previous history of cardiovascular and cerebrovascular diseases suddenly suffered from sharp chest pain and syncope after intensive exercise. The electrocardiogram showed that the ST segment of multiple leads was depressed by more than 0.05 mV, and biomarkers indicated severe myocardial injuries. The left ventricular ejection fraction (LVEF) decreased dramatically to 23%. Fulminant myocarditis and cardiomyopathy were therefore excluded. However, a relatively normal coronary artery origin, which arose from the left coronary sinus, presented on echocardiography and cardiac CT angiography (CTA). It is difficult to draw an association between severe clinical manifestations and slight malformations on echocardiography and CTA. Furthermore, selective coronary angiography revealed that an anomalous left coronary artery arose from the superior margin of the inappropriate sinus, developed an intramural wall course and finally exits the left sinus of Valsalva and migrated between the aorta and the pulmonary artery, which induced severe myocardial infarction during exercise. Then, the patient received surgical correction with a modified unroofing procedure. After 2 months of intensive treatment, the patient was discharged and remained asymptomatic through 18 months of follow-up. CONCLUSION: AAOCA, especially anomalous left coronary artery (ALCA), represents a major potential risk of SCD. We reported an atypical manifestation of ALCA arising from the inappropriate sinus of Valsalva and merging into the intra-arterial wall to develop a strange course and then sprout between the aorta and the pulmonary artery. The diversity of AAOCA might present as a relatively normal course under non-invasive radiological imaging scanning. Frontiers Media S.A. 2022-06-09 /pmc/articles/PMC9222707/ /pubmed/35757337 http://dx.doi.org/10.3389/fcvm.2022.918832 Text en Copyright © 2022 Hu, Wang, Wan, Li, Wang, Zhou, Shi, Liu, Fang and Hua. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Cardiovascular Medicine
Hu, Fan
Wang, Xinyue
Wan, Jiaojiao
Li, Yifei
Wang, Tao
Zhou, Kaiyu
Shi, Xiaoqing
Liu, Zhongqiang
Fang, Jie
Hua, Yimin
An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report
title An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report
title_full An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report
title_fullStr An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report
title_full_unstemmed An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report
title_short An Atypical Anomalous Aortic Origin of the Left Coronary Artery With Intra-Arterial Wall Course Pretending a Normal Migration on Imaging Screening: A Case Report
title_sort atypical anomalous aortic origin of the left coronary artery with intra-arterial wall course pretending a normal migration on imaging screening: a case report
topic Cardiovascular Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9222707/
https://www.ncbi.nlm.nih.gov/pubmed/35757337
http://dx.doi.org/10.3389/fcvm.2022.918832
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