Hepatocellular carcinoma and the Fontan circulation: Clinical presentation and outcomes.

Adult congenital heart disease Alpha-fetoprotein Fontan Fontan-associated liver disease Hepatocellular carcinoma Liver cirrhosis

Journal

International journal of cardiology
ISSN: 1874-1754
Titre abrégé: Int J Cardiol
Pays: Netherlands
ID NLM: 8200291

Informations de publication

Date de publication:
01 01 2021
Historique:
received: 04 06 2020
revised: 16 07 2020
accepted: 17 08 2020
pubmed: 24 8 2020
medline: 28 5 2021
entrez: 24 8 2020
Statut: ppublish

Résumé

Fontan-associated liver disease (FALD) is universal in patients with a Fontan circulation. Hepatocellular carcinoma (HCC) is one of its severe expressions, and, though rare, frequently fatal. The purpose of this study was to describe the clinical presentation, risk factors, and outcomes of HCC in patients with a Fontan circulation. A multicenter case series of Fontan patients with a diagnosis of HCC formed the basis of this study. The case series was extended by published cases and case reports. Clinical presentation, tumor characteristics, laboratory and hemodynamic findings as well as treatment types and outcomes, were described. Fifty-four Fontan patients (50% female) with a diagnosis of HCC were included. Mean age at HCC diagnosis was 30 ± 9.4 years and mean duration from Fontan surgery to HCC diagnosis was 21.6 ± 7.4 years. Median HCC size at the time of diagnosis was 4 cm with a range of 1 to 22 cm. The tumor was located in the right hepatic lobe in 65% of the patients. Fifty-one percent had liver cirrhosis at the time of HCC diagnosis. Fifty percent of the patients had no symptoms related to HCC and alpha-fetoprotein was normal in 26% of the cases. Twenty-six patients (48%) died during a median follow-up duration of 10.6 (range 1-50) months. HCC in Fontan patients occurs at a young age with a 1-year survival rate of only 50%. Meticulous liver surveillance is crucial to detect small tumors in the early stage.

Sections du résumé

BACKGROUND
Fontan-associated liver disease (FALD) is universal in patients with a Fontan circulation. Hepatocellular carcinoma (HCC) is one of its severe expressions, and, though rare, frequently fatal. The purpose of this study was to describe the clinical presentation, risk factors, and outcomes of HCC in patients with a Fontan circulation.
METHODS
A multicenter case series of Fontan patients with a diagnosis of HCC formed the basis of this study. The case series was extended by published cases and case reports. Clinical presentation, tumor characteristics, laboratory and hemodynamic findings as well as treatment types and outcomes, were described.
RESULTS
Fifty-four Fontan patients (50% female) with a diagnosis of HCC were included. Mean age at HCC diagnosis was 30 ± 9.4 years and mean duration from Fontan surgery to HCC diagnosis was 21.6 ± 7.4 years. Median HCC size at the time of diagnosis was 4 cm with a range of 1 to 22 cm. The tumor was located in the right hepatic lobe in 65% of the patients. Fifty-one percent had liver cirrhosis at the time of HCC diagnosis. Fifty percent of the patients had no symptoms related to HCC and alpha-fetoprotein was normal in 26% of the cases. Twenty-six patients (48%) died during a median follow-up duration of 10.6 (range 1-50) months.
CONCLUSIONS
HCC in Fontan patients occurs at a young age with a 1-year survival rate of only 50%. Meticulous liver surveillance is crucial to detect small tumors in the early stage.

Identifiants

pubmed: 32828959
pii: S0167-5273(20)33640-8
doi: 10.1016/j.ijcard.2020.08.057
pii:
doi:

Types de publication

Journal Article Multicenter Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

142-148

Informations de copyright

Crown Copyright © 2020. Published by Elsevier B.V. All rights reserved.

Auteurs

Mathias Possner (M)

Division of Cardiology, Department of Medicine, University of Washington, Seattle, Washington, USA.

Timothy Gordon-Walker (T)

Scottish Liver Transplant Unit, Royal Infirmary of Edinburgh, Edinburgh, United Kingdom; The University of Edinburgh, Edinburgh, United Kingdom.

Alexander C Egbe (AC)

Department of Cardiovascular Medicine, Mayo Clinic, Rochester, MN, USA.

Joseph T Poterucha (JT)

Department of Cardiovascular Medicine, Mayo Clinic, Rochester, MN, USA.

Carole A Warnes (CA)

Department of Cardiovascular Medicine, Mayo Clinic, Rochester, MN, USA.

Heidi M Connolly (HM)

Department of Cardiovascular Medicine, Mayo Clinic, Rochester, MN, USA.

Salil Ginde (S)

Division of Pediatric Cardiology, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, USA.

Paul Clift (P)

Department of Cardiology, New Queen Elizabeth Hospital, Birmingham, United Kingdom.

Brian Kogon (B)

Department of Cardiothoracic Surgery, Children's Healthcare of Atlanta, Egleston, Emory University, Atlanta, GA, USA.

Wendy M Book (WM)

Division of Cardiology, Department of Medicine, Emory University Department of Medicine, Atlanta, GA, USA.

Niki Walker (N)

Scottish Adult Congenital Cardiac Service, Golden Jubilee National Hospital, Glasgow, United Kingdom.

Lodewijk J Wagenaar (LJ)

Department of Cardiology, Thoraxcentrum Twente, Medisch Spectrum Twente Hospital, Enschede, the Netherlands.

Tabitha Moe (T)

Division of Cardiology, Phoenix Children's Hospital, Phoenix, AZ, USA.

Erwin Oechslin (E)

Toronto Congenital Cardiac Centre for Adults, University Health Network / Peter Munk Cardiac Centre, and University of Toronto, Toronto, Ontario, Canada.

W Aaron Kay (WA)

Section of Pediatric Cardiology, Department of Pediatrics, Indiana University School of Medicine, Indianapolis, Indiana, USA; Krannert Institute of Cardiology, Department of Medicine, Indiana University School of Medicine, Indianapolis, Indiana, USA.

Mark Norris (M)

Division of Pediatric Cardiology, University of Michigan Congenital Heart Center, C.S. Mott Children's Hospital, Ann Arbor, MI, USA.

Jonathan R Dillman (JR)

Department of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA; Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.

Andrew T Trout (AT)

Department of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA; Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.

Nadeem Anwar (N)

Division of Gastroenterology, Department of Medicine, University of Cincinnati College of Medicine, Cincinnati, OH, USA.

Arvind Hoskoppal (A)

Department of Pediatrics, University of Utah and Intermountain Healthcare, Salt Lake City, UT, USA.

Dieter C Broering (DC)

Department of Liver Transplantation, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia.

Khalid Bzeizi (K)

Adult Congenital Heart Disease, Heart Centre, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia.

Gruschen Veldtman (G)

Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA; Adult Congenital Heart Disease, Heart Centre, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia. Electronic address: Gruschen@me.com.

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