Diagnosis and Surgical Outcomes of Patients with Anomalous Left Coronary Artery from the Pulmonary Artery: A Single Taiwanese Medical Center Experience.


Journal

The heart surgery forum
ISSN: 1522-6662
Titre abrégé: Heart Surg Forum
Pays: United States
ID NLM: 100891112

Informations de publication

Date de publication:
05 03 2020
Historique:
received: 07 08 2019
accepted: 04 12 2019
entrez: 5 5 2020
pubmed: 5 5 2020
medline: 5 1 2021
Statut: epublish

Résumé

Anomalous left coronary artery from the pulmonary artery (ALCAPA), a very rare congenital cardiac anomaly, is associated with a high mortality rate among infants who are not diagnosed or treated in a timely manner. Surgical intervention with the reconstruction for a two-coronary-system circulation is the main treatment; however, there have been very few reported cases from Taiwan. In this study, we aim to describe the clinical manifestations, diagnostic methods, surgery types, and surgical outcomes in patients with ALCAPA from a single Taiwanese medical center. We retrospectively reviewed patients diagnosed with ALCAPA who underwent surgery at our institution between January 2001 and October 2018. Clinical presentations, noninvasive and invasive study results, surgical methods, and postoperative follow-up results were assessed from medical records. Moreover, literature on this particular cardiovascular anomaly was reviewed. The study included 6 patients (5 children and 1 adult). The diagnosis was confirmed using cardiac catheterization and coronary angiography in four patients and only echocardiography in two patients. All patients underwent surgical correction and survived. Four patients showed improvements in left ventricular function and mitral regurgitation (MR). Early diagnosis and timely surgical intervention could avoid mortality regardless of the method of operation. ALCAPA can be definitively diagnosed using noninvasive echocardiography. Both left ventricular systolic function and mitral insufficiency could improve after the surgical intervention in pediatric patients. Repair or replacement of the mitral valve could be reserved for persistent MR complicated with congestive heart failure, particularly in patients who received the initial operation beyond infancy.

Sections du résumé

BACKGROUND
Anomalous left coronary artery from the pulmonary artery (ALCAPA), a very rare congenital cardiac anomaly, is associated with a high mortality rate among infants who are not diagnosed or treated in a timely manner. Surgical intervention with the reconstruction for a two-coronary-system circulation is the main treatment; however, there have been very few reported cases from Taiwan. In this study, we aim to describe the clinical manifestations, diagnostic methods, surgery types, and surgical outcomes in patients with ALCAPA from a single Taiwanese medical center.
METHODS
We retrospectively reviewed patients diagnosed with ALCAPA who underwent surgery at our institution between January 2001 and October 2018. Clinical presentations, noninvasive and invasive study results, surgical methods, and postoperative follow-up results were assessed from medical records. Moreover, literature on this particular cardiovascular anomaly was reviewed.
RESULTS
The study included 6 patients (5 children and 1 adult). The diagnosis was confirmed using cardiac catheterization and coronary angiography in four patients and only echocardiography in two patients. All patients underwent surgical correction and survived. Four patients showed improvements in left ventricular function and mitral regurgitation (MR).
CONCLUSION
Early diagnosis and timely surgical intervention could avoid mortality regardless of the method of operation. ALCAPA can be definitively diagnosed using noninvasive echocardiography. Both left ventricular systolic function and mitral insufficiency could improve after the surgical intervention in pediatric patients. Repair or replacement of the mitral valve could be reserved for persistent MR complicated with congestive heart failure, particularly in patients who received the initial operation beyond infancy.

Identifiants

pubmed: 32364892
doi: 10.1532/hsf.2703
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

E101-E106

Informations de copyright

2020 Forum Multimedia Publishing, LLC

Auteurs

Wan-Fu Hsu (WF)

Department of Pediatrics, Tri-Service General Hospital, National Defense Medical Center, Taipei City, Taiwan, ROC.
Department of Pediatrics, Penghu Branch of Tri-Service General Hospital, National Defense Medical Center, Penghu County, Taiwan, ROC.
Division of Pediatric Cardiology, Children’s Medical Center, Taipei Veterans General Hospital, Taipei City, Taiwan, ROC.

Pi-Chang Lee (PC)

Division of Pediatric Cardiology, Children’s Medical Center, Taipei Veterans General Hospital, Taipei City, Taiwan, ROC.
School of Medicine, National Yang-Ming University, Taipei City, Taiwan, ROC.

Hsing-Yuan Li (HY)

Division of Pediatric Cardiology, Children’s Medical Center, Taipei Veterans General Hospital, Taipei City, Taiwan, ROC.
School of Medicine, National Yang-Ming University, Taipei City, Taiwan, ROC.

Shao-Wei Huang (SW)

Department of Pediatrics, Tri-Service General Hospital, National Defense Medical Center, Taipei City, Taiwan, ROC.
Department of Pediatrics, Penghu Branch of Tri-Service General Hospital, National Defense Medical Center, Penghu County, Taiwan, ROC.
Division of Pediatric Cardiology, Children’s Medical Center, Taipei Veterans General Hospital, Taipei City, Taiwan, ROC.

Shyi-Jou Chen (SJ)

Department of Pediatrics, Tri-Service General Hospital, National Defense Medical Center, Taipei City, Taiwan, ROC.

Fei-Yi Wu (FY)

School of Medicine, National Yang-Ming University, Taipei City, Taiwan, ROC.
Division of Cardiovascular Surgery, Department of Surgery, Taipei Veterans General Hospital, Taipei City, Taiwan, ROC.

Zen-Chung Weng (ZC)

Division of Cardiovascular Surgery, Department of Surgery, School of Medicine, College of Medicine, Taipei Medical University Hospital, Taipei City, Taiwan, ROC

Be Tau Hwang (BT)

School of Medicine, National Yang-Ming University, Taipei City, Taiwan, ROC.
Department of Pediatrics, Tungs’ Taichung MetroHarbor Hospital, Taichung City, Taiwan, ROC.

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