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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

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 admitte...

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Autores principales: Hölscher, Martin, Knirsch, Walter, Dave, Hitendu, Burkhardt, Barbara E U
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6939823/
https://www.ncbi.nlm.nih.gov/pubmed/31912005
http://dx.doi.org/10.1093/ehjcr/ytz166
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author Hölscher, Martin
Knirsch, Walter
Dave, Hitendu
Burkhardt, Barbara E U
author_facet Hölscher, Martin
Knirsch, Walter
Dave, Hitendu
Burkhardt, Barbara E U
author_sort Hölscher, Martin
collection PubMed
description 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.
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spelling pubmed-69398232020-01-07 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 Hölscher, Martin Knirsch, Walter Dave, Hitendu Burkhardt, Barbara E U Eur Heart J Case Rep Case Reports 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. Oxford University Press 2019-10-08 /pmc/articles/PMC6939823/ /pubmed/31912005 http://dx.doi.org/10.1093/ehjcr/ytz166 Text en © The Author(s) 2019. Published by Oxford University Press on behalf of the European Society of Cardiology. http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Case Reports
Hölscher, Martin
Knirsch, Walter
Dave, Hitendu
Burkhardt, Barbara E U
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
title 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
title_full 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
title_fullStr 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
title_full_unstemmed 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
title_short 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
title_sort 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
topic Case Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6939823/
https://www.ncbi.nlm.nih.gov/pubmed/31912005
http://dx.doi.org/10.1093/ehjcr/ytz166
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