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患有主动脉缩窄及左冠状动脉起源于肺动脉并伴有左旋支动脉逆行灌注的新生女婴:一例报告

Newborn girl with coarctation of the aorta and anomalous left coronary artery from pulmonary artery, with retrograde perfusion of left circumflex artery: a case report.

作者信息

Hölscher Martin, Knirsch Walter, Dave Hitendu, Burkhardt Barbara E U

机构信息

Pediatric Cardiology, Department of Surgery, Pediatric Heart Center, University Children's Hospital Zurich, Steinwiesstrasse 75, 8032 Zurich, Switzerland.

Department of Surgery, Pediatric Cardiovascular Surgery, Pediatric Heart Center, University Children's Hospital Zurich, Steinwiesstrasse 75, 8032 Zurich, Switzerland.

出版信息

Eur Heart J Case Rep. 2019 Dec;3(4):1-4. doi: 10.1093/ehjcr/ytz166. Epub 2019 Oct 8.

DOI:10.1093/ehjcr/ytz166
PMID:31912005
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6939823/
Abstract

BACKGROUND

Anomalous left coronary artery from pulmonary artery (ALCAPA) is a rare coronary abnormality. Although it exists usually as an isolated abnormality, ALCAPA has been described with aortic pathologies like coarctation or aortopulmonary window.

CASE SUMMARY

An 18-day-old female was admitted to the paediatric intensive care unit because of a heart murmur and weak femoral pulses. A transthoracic two-dimensional echocardiography was performed and confirmed suspected diagnosis of aortic coarctation. In addition, a total retrograde perfusion of the left circumflex coronary artery (LCX) was found, without visible flow through the ostium of the left coronary artery (LCA) into the aorta. A coronary angiography was performed, showing a single right coronary artery with a normal right posterior descending artery (RPD). Supplied by collaterals from the RPD, the LCX was perfused retrogradely, passing by the lateral wall of the ascending aorta without flowing into it, but into the right pulmonary artery. At 23 days of age, surgery was performed with resection of the aortic coarctation and reimplantation of the LCA into the posterior aortic wall.

DISCUSSION

This case demonstrates that coronary artery anomalies like ALCAPA may occur together with other cardiac malformations. Despite concomitant cardiac lesions, careful assessment of the coronary arteries is mandatory, including cardiac catheterization in case of doubt.

摘要

背景

起源于肺动脉的异常左冠状动脉(ALCAPA)是一种罕见的冠状动脉异常。虽然它通常作为一种孤立的异常存在,但ALCAPA已被描述与诸如主动脉缩窄或主肺动脉窗等主动脉病变相关。

病例摘要

一名18日龄女性因心脏杂音和股动脉搏动减弱入住儿科重症监护病房。进行了经胸二维超声心动图检查,证实了疑似主动脉缩窄的诊断。此外,发现左旋支冠状动脉(LCX)完全逆行灌注,未见左冠状动脉(LCA)开口处有血流进入主动脉。进行了冠状动脉造影,显示单支右冠状动脉伴正常的右后降支(RPD)。由RPD的侧支供血,LCX逆行灌注,经过升主动脉侧壁但未流入升主动脉,而是流入右肺动脉。在23日龄时,进行了手术,切除主动脉缩窄并将LCA重新植入主动脉后壁。

讨论

本病例表明,像ALCAPA这样的冠状动脉异常可能与其他心脏畸形同时发生。尽管存在合并的心脏病变,但必须仔细评估冠状动脉,如有疑问包括进行心导管检查。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/3435cece8837/ytz166f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/ff1c31c0a8bf/ytz166f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/9b36f418ed9c/ytz166f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/868f37aef86c/ytz166f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/3435cece8837/ytz166f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/ff1c31c0a8bf/ytz166f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/9b36f418ed9c/ytz166f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/868f37aef86c/ytz166f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dc39/6939823/3435cece8837/ytz166f4.jpg

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本文引用的文献

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Clinical guidelines for the management of patients with transposition of the great arteries with intact ventricular septum.室间隔完整型大动脉转位患者管理的临床指南
Eur J Cardiothorac Surg. 2017 Jan;51(1):e1-e32. doi: 10.1093/ejcts/ezw360.
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Anomalous origin of circumflex coronary artery from right pulmonary artery associated with coarctation of the aorta: a case report of surgical treatment.右肺动脉发出回旋支冠状动脉异常并合并主动脉缩窄:手术治疗病例报告
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Anomalous right coronary artery arising from the pulmonary artery: a report of 7 cases and a review of the literature.
起源于肺动脉的右冠状动脉异常:7例报告并文献复习
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Origin of the circumflex coronary artery from the pulmonary artery in infants.婴儿冠状动脉回旋支起源于肺动脉的情况。
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