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Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report

BACKGROUND: Anomalous coronary origin from the left ventricular outflow tract (LVOT) is an exceedingly rare condition thought to be associated with the bicuspid aortic valve (BAV). While the malignant presentation of this entity has been described, its pathophysiology and diagnostic evaluation are p...

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Autores principales: Venkatesh, Prashanth, Tan, Weiyi, Bravo-Jaimes, Katia, Aboulhosn, Jamil
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9336575/
https://www.ncbi.nlm.nih.gov/pubmed/35911489
http://dx.doi.org/10.1093/ehjcr/ytac237
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author Venkatesh, Prashanth
Tan, Weiyi
Bravo-Jaimes, Katia
Aboulhosn, Jamil
author_facet Venkatesh, Prashanth
Tan, Weiyi
Bravo-Jaimes, Katia
Aboulhosn, Jamil
author_sort Venkatesh, Prashanth
collection PubMed
description BACKGROUND: Anomalous coronary origin from the left ventricular outflow tract (LVOT) is an exceedingly rare condition thought to be associated with the bicuspid aortic valve (BAV). While the malignant presentation of this entity has been described, its pathophysiology and diagnostic evaluation are poorly understood. CASE SUMMARY: A 33-year-old woman status post Ross procedure in childhood for congenital aortic stenosis due to BAV with presumed common origin of right and left coronary arteries based on single coronary ostium seen on aortic valve inspection, presented with symptomatic pulmonary regurgitation and stenosis. Invasive left coronary angiography revealed retrograde filling of the right coronary artery (RCA) with systolic washout of contrast indicating a patent RCA ostium. No RCA ostium was found on aortic root injection, but an injection into the LVOT revealed an RCA ostium below the aortic valve. Selective RCA angiography revealed pulsatile antegrade flow down the RCA occurring during systole. There was no anatomic RCA stenosis. We proceeded with valve-in-valve TcPVR. The patient had significant improvement of symptoms and RCA reimplantation was hence deferred. DISCUSSION: This case is the first of an anomalous coronary artery arising from the LVOT diagnosed in a patient after the Ross procedure. Our angiograms shed light on the unusual physiology of coronary filling during systole and ischaemia arising from inadequate perfusion gradient between the left ventricle and the coronary during systole, leading to collateralization despite the lack of anatomic stenosis. We urge consideration of this potentially malignant entity in any symptomatic patient, especially with concomitant BAV.
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spelling pubmed-93365752022-07-29 Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report Venkatesh, Prashanth Tan, Weiyi Bravo-Jaimes, Katia Aboulhosn, Jamil Eur Heart J Case Rep Case Report BACKGROUND: Anomalous coronary origin from the left ventricular outflow tract (LVOT) is an exceedingly rare condition thought to be associated with the bicuspid aortic valve (BAV). While the malignant presentation of this entity has been described, its pathophysiology and diagnostic evaluation are poorly understood. CASE SUMMARY: A 33-year-old woman status post Ross procedure in childhood for congenital aortic stenosis due to BAV with presumed common origin of right and left coronary arteries based on single coronary ostium seen on aortic valve inspection, presented with symptomatic pulmonary regurgitation and stenosis. Invasive left coronary angiography revealed retrograde filling of the right coronary artery (RCA) with systolic washout of contrast indicating a patent RCA ostium. No RCA ostium was found on aortic root injection, but an injection into the LVOT revealed an RCA ostium below the aortic valve. Selective RCA angiography revealed pulsatile antegrade flow down the RCA occurring during systole. There was no anatomic RCA stenosis. We proceeded with valve-in-valve TcPVR. The patient had significant improvement of symptoms and RCA reimplantation was hence deferred. DISCUSSION: This case is the first of an anomalous coronary artery arising from the LVOT diagnosed in a patient after the Ross procedure. Our angiograms shed light on the unusual physiology of coronary filling during systole and ischaemia arising from inadequate perfusion gradient between the left ventricle and the coronary during systole, leading to collateralization despite the lack of anatomic stenosis. We urge consideration of this potentially malignant entity in any symptomatic patient, especially with concomitant BAV. Oxford University Press 2022-06-15 /pmc/articles/PMC9336575/ /pubmed/35911489 http://dx.doi.org/10.1093/ehjcr/ytac237 Text en © The Author(s) 2022. Published by Oxford University Press on behalf of the European Society of Cardiology. https://creativecommons.org/licenses/by-nc/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial License (https://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 Report
Venkatesh, Prashanth
Tan, Weiyi
Bravo-Jaimes, Katia
Aboulhosn, Jamil
Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report
title Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report
title_full Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report
title_fullStr Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report
title_full_unstemmed Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report
title_short Right coronary artery originating from the left ventricular outflow tract diagnosed after a Ross procedure: a case report
title_sort right coronary artery originating from the left ventricular outflow tract diagnosed after a ross procedure: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9336575/
https://www.ncbi.nlm.nih.gov/pubmed/35911489
http://dx.doi.org/10.1093/ehjcr/ytac237
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