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Total anomalous origin of the coronary arteries from the pulmonary artery: a systematic review

Published online by Cambridge University Press:  26 July 2021

Timothy M. Guenther*
Affiliation:
Department of Surgery, University of California Davis, 2335 Stockton Blvd, SacramentoCA95817, USA Department of Cardiothoracic Surgery, David Grant USAF Medical Center, 101 Bolin Circle, Travis AFB, CA95433, USA
Sarah A. Chen
Affiliation:
Department of Surgery, University of California Davis, 2335 Stockton Blvd, SacramentoCA95817, USA
Joshua D. Gustafson
Affiliation:
Department of Cardiothoracic Surgery, David Grant USAF Medical Center, 101 Bolin Circle, Travis AFB, CA95433, USA
Frank F. Ing
Affiliation:
Department of Pediatrics, University of California Davis, 2335 Stockton Blvd, SacramentoCA95817, USA
Julie A. Brothers
Affiliation:
Department of Cardiology, The Children’s Hospital of Philadelphia, 3401 Civic Center Blvd, Philadelphia, PA19104, USA
Gary W. Raff
Affiliation:
Department of Surgery, University of California Davis, 2335 Stockton Blvd, SacramentoCA95817, USA
*
Author for correspondence: T. M. Guenther, MD, 2335 Stockton Blvd, North Addition 5th floor, Sacramento, CA95817, USA. Tel: +916-734-3528; Fax: 916-734-3951. E-mail: guenther.tim@gmail.com.

Abstract

Background:

Although rare, coronary artery anomalies can have significant clinical implications. Total anomalous origin of the coronary arteries from the pulmonary artery (TCAPA) represents a rare subtype of coronary artery anomaly for which little is known. The aim of this review was to characterise the presentation, utilised diagnostic modalities, associated cardiac lesions, and treatment strategies in patients with TCAPA.

Methods:

A systematic review was performed for cases of TCAPA using PubMed, Embase, and Web of Science. Keywords searched included “total anomalous origin of the coronary arteries from the pulmonary artery,” “single ostium anomalous coronary artery from the pulmonary artery,” and “anomalous origin of both coronary arteries from the pulmonary artery.”

Results:

Fifty-seven cases of TCAPA were identified in 50 manuscripts. Fifty-eight per cent of patients were male and the median age at presentation was 10 days (mean 1.71 ± 6.6 years, range 0 days–39 years). Most patients were symptomatic at the time of presentation; cyanosis (n = 22) and respiratory distress (n = 14) were the most common symptoms. Cases were most commonly diagnosed at autopsy (n = 26, 45.6%), but operative intervention was pursued in 22 cases (45.6%); aortic re-implantation (n = 14) and a Takeuchi-type repair (n = 7) were the most common routes of repair.

Conclusions:

The clinical presentation of patients with TCAPA was found to be variable, likely related to the presence of associated cardiac lesions. TCAPA should be considered in patients with suspected anomalous origin of the left coronary artery from the pulmonary artery for the serious consequences that can occur if not promptly corrected.

Type
Review
Copyright
© The Author(s), 2021. Published by Cambridge University Press

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Footnotes

This work was not presented at any scientific meetings

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