A Rare Report of Hypoplastic Coronary Arteries and Pulmonary Veins: A Case Report and Review of the Literature
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CASE REPORT
A Rare Report of Hypoplastic Coronary Arteries and Pulmonary Veins: A Case Report and Review of the Literature Raymond Morales1 · John Bokowski1 · Saif Aljemmali1 · Joshua Murphy1 · Saroja Bharati2,3,4,5 · Ra‑id Abdulla6 Received: 9 January 2020 / Accepted: 21 February 2020 © Springer Science+Business Media, LLC, part of Springer Nature 2020
Abstract Congenital coronary artery anomalies are estimated to affect about 1% of the general population. Hypoplastic coronary artery disease is an uncommon subset associated with significant mortality regardless whether another major cardiac anomaly is present. In this case report, we present an extremely rare case of an infant with hypoplastic coronary artery disease and hypoplastic pulmonary veins. A literature review pertinent to the clinical findings is also contained herein. Keywords Hypoplastic coronary artery disease · Pulmonary vein stenosis · Diminutive coronary artery syndrome
Introduction Congenital coronary artery anomalies consist of a heterogeneous group of malformations that affect 1% of the general population [1]. Hypoplastic coronary artery disease (HCAD) is a rare subset that was first described in a pathology report of an adolescent female with a rudimentary right coronary artery who died from acute appendicitis complicated by abscess formation [2]. Postmortem on a 17-year-old female was the first reported case of a sudden cardiac death event attributed to a hypoplastic right coronary artery while postmortem on a 38-year-old woman was the first case attributed to a hypoplastic left coronary artery [3, 4]. HCAD is defined Electronic supplementary material The online version of this article (https://doi.org/10.1007/s00246-020-02334-x) contains supplementary material, which is available to authorized users. * Raymond Morales [email protected] 1
Division of Pediatric Cardiology, Department of Pediatrics, Rush University Medical Center, Chicago, IL, USA
2
Department of Pathology, Rush University Medical Center, Chicago, IL, USA
3
Rosalind Franklin University of Sciences, North Chicago, IL, USA
4
The Advocate Children’s Hospital, Oak Lawn, IL, USA
5
Advocate Aurora Healthcare, Aurora, IL, USA
6
Pediatric Cardiology, Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, IL, USA
as a small luminal diameter for age of at least one coronary artery or major coronary branch distal to the ostia [5]. The term diminutive coronary artery syndrome (DCAS) has been proposed for HCAD patients who manifest clinically with myocardial ischemia [6]. With that said, it has been known for some time that patients with HCAD are at high risk for ischemic events and that sudden cardiac death is the most common form of presentation [7]. In this case report, we document the course of a term infant with HCAD and an even rarer combination of hypoplastic pulmonary veins.
Case Report Patient was a female diamniotic-dichorionic twin B born small-for-gestational-age (birth weight 1540 g) at 37 2/7 weeks gestati
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