Longitudinal Outcomes of Patients With Single Ventricle After the Fontan Procedure

Multicenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking. This study sought to describe transplant-free survival and explore relationships between laboratory measures of ventricular performance and functional status over time. E...

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Veröffentlicht in:Journal of the American College of Cardiology Jg. 69; H. 22; S. 2735
Hauptverfasser: Atz, Andrew M, Zak, Victor, Mahony, Lynn, Uzark, Karen, D'agincourt, Nicholas, Goldberg, David J, Williams, Richard V, Breitbart, Roger E, Colan, Steven D, Burns, Kristin M, Margossian, Renee, Henderson, Heather T, Korsin, Rosalind, Marino, Bradley S, Daniels, Kaitlyn, McCrindle, Brian W
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Sprache:Englisch
Veröffentlicht: United States 06.06.2017
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ISSN:1558-3597, 1558-3597
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Abstract Multicenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking. This study sought to describe transplant-free survival and explore relationships between laboratory measures of ventricular performance and functional status over time. Exercise testing, echocardiography, B-type natriuretic peptide, functional health assessment, and medical history abstraction were repeated 9.4 ± 0.4 years after the Fontan Cross-Sectional Study (Fontan 1) and compared with previous values. Cox regression analysis explored risk factors for interim death or cardiac transplantation. From the original cohort of 546 subjects, 466 were contacted again, and 373 (80%) were enrolled at 21.2 ± 3.5 years of age. Among subjects with paired testing, the percent predicted maximum oxygen uptake decreased (69 ± 14% vs. 61 ± 16%; p < 0.001; n = 95), ejection fraction decreased (58 ± 11% vs. 55 ± 10%; p < 0.001; n = 259), and B-type natriuretic peptide increased (median [interquartile range] 13 [7 to 25] pg/mol vs. 18 [9 to 36] pg/mol; p < 0.001; n = 340). At latest follow-up, a lower Pediatric Quality of Life Inventory physical summary score was associated with poorer exercise performance (R adjusted = 0.20; p < 0.001; n = 274). Cumulative complications since the Fontan procedure included additional cardiac surgery (32%), catheter intervention (62%), arrhythmia treatment (32%), thrombosis (12%), and protein-losing enteropathy (8%). Since Fontan 1, 54 subjects (10%) have received a heart transplant (n = 23) or died without transplantation (n = 31). The interval risk of death or/cardiac transplantation was associated with poorer ventricular performance and functional health status assessed at Fontan 1, but it was not associated with ventricular morphology, the subject's age, or the type of Fontan connection. Interim transplant-free survival over 12 years in this Fontan cohort was 90% and was independent of ventricular morphology. Exercise performance decreased and was associated with worse functional health status. Future interventions might focus on preserving exercise capacity. (Relationship Between Functional Health Status and Ventricular Performance After Fontan-Pediatric Heart Network; NCT00132782).
AbstractList Multicenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking. This study sought to describe transplant-free survival and explore relationships between laboratory measures of ventricular performance and functional status over time. Exercise testing, echocardiography, B-type natriuretic peptide, functional health assessment, and medical history abstraction were repeated 9.4 ± 0.4 years after the Fontan Cross-Sectional Study (Fontan 1) and compared with previous values. Cox regression analysis explored risk factors for interim death or cardiac transplantation. From the original cohort of 546 subjects, 466 were contacted again, and 373 (80%) were enrolled at 21.2 ± 3.5 years of age. Among subjects with paired testing, the percent predicted maximum oxygen uptake decreased (69 ± 14% vs. 61 ± 16%; p < 0.001; n = 95), ejection fraction decreased (58 ± 11% vs. 55 ± 10%; p < 0.001; n = 259), and B-type natriuretic peptide increased (median [interquartile range] 13 [7 to 25] pg/mol vs. 18 [9 to 36] pg/mol; p < 0.001; n = 340). At latest follow-up, a lower Pediatric Quality of Life Inventory physical summary score was associated with poorer exercise performance (R adjusted = 0.20; p < 0.001; n = 274). Cumulative complications since the Fontan procedure included additional cardiac surgery (32%), catheter intervention (62%), arrhythmia treatment (32%), thrombosis (12%), and protein-losing enteropathy (8%). Since Fontan 1, 54 subjects (10%) have received a heart transplant (n = 23) or died without transplantation (n = 31). The interval risk of death or/cardiac transplantation was associated with poorer ventricular performance and functional health status assessed at Fontan 1, but it was not associated with ventricular morphology, the subject's age, or the type of Fontan connection. Interim transplant-free survival over 12 years in this Fontan cohort was 90% and was independent of ventricular morphology. Exercise performance decreased and was associated with worse functional health status. Future interventions might focus on preserving exercise capacity. (Relationship Between Functional Health Status and Ventricular Performance After Fontan-Pediatric Heart Network; NCT00132782).
Multicenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking.BACKGROUNDMulticenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking.This study sought to describe transplant-free survival and explore relationships between laboratory measures of ventricular performance and functional status over time.OBJECTIVESThis study sought to describe transplant-free survival and explore relationships between laboratory measures of ventricular performance and functional status over time.Exercise testing, echocardiography, B-type natriuretic peptide, functional health assessment, and medical history abstraction were repeated 9.4 ± 0.4 years after the Fontan Cross-Sectional Study (Fontan 1) and compared with previous values. Cox regression analysis explored risk factors for interim death or cardiac transplantation.METHODSExercise testing, echocardiography, B-type natriuretic peptide, functional health assessment, and medical history abstraction were repeated 9.4 ± 0.4 years after the Fontan Cross-Sectional Study (Fontan 1) and compared with previous values. Cox regression analysis explored risk factors for interim death or cardiac transplantation.From the original cohort of 546 subjects, 466 were contacted again, and 373 (80%) were enrolled at 21.2 ± 3.5 years of age. Among subjects with paired testing, the percent predicted maximum oxygen uptake decreased (69 ± 14% vs. 61 ± 16%; p < 0.001; n = 95), ejection fraction decreased (58 ± 11% vs. 55 ± 10%; p < 0.001; n = 259), and B-type natriuretic peptide increased (median [interquartile range] 13 [7 to 25] pg/mol vs. 18 [9 to 36] pg/mol; p < 0.001; n = 340). At latest follow-up, a lower Pediatric Quality of Life Inventory physical summary score was associated with poorer exercise performance (R2 adjusted = 0.20; p < 0.001; n = 274). Cumulative complications since the Fontan procedure included additional cardiac surgery (32%), catheter intervention (62%), arrhythmia treatment (32%), thrombosis (12%), and protein-losing enteropathy (8%). Since Fontan 1, 54 subjects (10%) have received a heart transplant (n = 23) or died without transplantation (n = 31). The interval risk of death or/cardiac transplantation was associated with poorer ventricular performance and functional health status assessed at Fontan 1, but it was not associated with ventricular morphology, the subject's age, or the type of Fontan connection.RESULTSFrom the original cohort of 546 subjects, 466 were contacted again, and 373 (80%) were enrolled at 21.2 ± 3.5 years of age. Among subjects with paired testing, the percent predicted maximum oxygen uptake decreased (69 ± 14% vs. 61 ± 16%; p < 0.001; n = 95), ejection fraction decreased (58 ± 11% vs. 55 ± 10%; p < 0.001; n = 259), and B-type natriuretic peptide increased (median [interquartile range] 13 [7 to 25] pg/mol vs. 18 [9 to 36] pg/mol; p < 0.001; n = 340). At latest follow-up, a lower Pediatric Quality of Life Inventory physical summary score was associated with poorer exercise performance (R2 adjusted = 0.20; p < 0.001; n = 274). Cumulative complications since the Fontan procedure included additional cardiac surgery (32%), catheter intervention (62%), arrhythmia treatment (32%), thrombosis (12%), and protein-losing enteropathy (8%). Since Fontan 1, 54 subjects (10%) have received a heart transplant (n = 23) or died without transplantation (n = 31). The interval risk of death or/cardiac transplantation was associated with poorer ventricular performance and functional health status assessed at Fontan 1, but it was not associated with ventricular morphology, the subject's age, or the type of Fontan connection.Interim transplant-free survival over 12 years in this Fontan cohort was 90% and was independent of ventricular morphology. Exercise performance decreased and was associated with worse functional health status. Future interventions might focus on preserving exercise capacity. (Relationship Between Functional Health Status and Ventricular Performance After Fontan-Pediatric Heart Network; NCT00132782).CONCLUSIONSInterim transplant-free survival over 12 years in this Fontan cohort was 90% and was independent of ventricular morphology. Exercise performance decreased and was associated with worse functional health status. Future interventions might focus on preserving exercise capacity. (Relationship Between Functional Health Status and Ventricular Performance After Fontan-Pediatric Heart Network; NCT00132782).
Author Margossian, Renee
McCrindle, Brian W
Daniels, Kaitlyn
Marino, Bradley S
Breitbart, Roger E
Williams, Richard V
Goldberg, David J
Henderson, Heather T
Mahony, Lynn
Burns, Kristin M
Korsin, Rosalind
Atz, Andrew M
Zak, Victor
D'agincourt, Nicholas
Colan, Steven D
Uzark, Karen
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  surname: Atz
  fullname: Atz, Andrew M
  email: atzam@musc.edu
  organization: Medical University of South Carolina, Charleston, South Carolina. Electronic address: atzam@musc.edu
– sequence: 2
  givenname: Victor
  surname: Zak
  fullname: Zak, Victor
  organization: New England Research Institutes, Watertown, Massachusetts
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  givenname: Lynn
  surname: Mahony
  fullname: Mahony, Lynn
  organization: University of Texas Southwestern Medical Center, Dallas, Texas
– sequence: 4
  givenname: Karen
  surname: Uzark
  fullname: Uzark, Karen
  organization: University of Michigan, Ann Arbor, Michigan
– sequence: 5
  givenname: Nicholas
  surname: D'agincourt
  fullname: D'agincourt, Nicholas
  organization: New England Research Institutes, Watertown, Massachusetts
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  surname: Goldberg
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  organization: Children's Hospital of Philadelphia, Philadelphia, Pennsylvania
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  givenname: Richard V
  surname: Williams
  fullname: Williams, Richard V
  organization: University of Utah, Salt Lake City, Utah
– sequence: 8
  givenname: Roger E
  surname: Breitbart
  fullname: Breitbart, Roger E
  organization: Boston Children's Hospital, Boston, Massachusetts
– sequence: 9
  givenname: Steven D
  surname: Colan
  fullname: Colan, Steven D
  organization: Boston Children's Hospital, Boston, Massachusetts
– sequence: 10
  givenname: Kristin M
  surname: Burns
  fullname: Burns, Kristin M
  organization: National Heart, Lung, and Blood Institute, Bethesda, Maryland
– sequence: 11
  givenname: Renee
  surname: Margossian
  fullname: Margossian, Renee
  organization: Boston Children's Hospital, Boston, Massachusetts
– sequence: 12
  givenname: Heather T
  surname: Henderson
  fullname: Henderson, Heather T
  organization: Duke University Medical Center, Durham, North Carolina
– sequence: 13
  givenname: Rosalind
  surname: Korsin
  fullname: Korsin, Rosalind
  organization: Columbia University Medical Center, New York, New York
– sequence: 14
  givenname: Bradley S
  surname: Marino
  fullname: Marino, Bradley S
  organization: Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio
– sequence: 15
  givenname: Kaitlyn
  surname: Daniels
  fullname: Daniels, Kaitlyn
  organization: University of Utah, Salt Lake City, Utah
– sequence: 16
  givenname: Brian W
  surname: McCrindle
  fullname: McCrindle, Brian W
  organization: University of Toronto, The Hospital for Sick Children, Toronto, Ontario, Canada
BackLink https://www.ncbi.nlm.nih.gov/pubmed/28571639$$D View this record in MEDLINE/PubMed
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ContentType Journal Article
Copyright Copyright © 2017 American College of Cardiology Foundation. Published by Elsevier Inc. All rights reserved.
Copyright_xml – notice: Copyright © 2017 American College of Cardiology Foundation. Published by Elsevier Inc. All rights reserved.
CorporateAuthor Pediatric Heart Network Investigators
CorporateAuthor_xml – name: Pediatric Heart Network Investigators
DBID CGR
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EIF
NPM
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DOI 10.1016/j.jacc.2017.03.582
DatabaseName Medline
MEDLINE
MEDLINE (Ovid)
MEDLINE
MEDLINE
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Issue 22
Keywords functional health status
exercise
Fontan procedure
adult congenital heart disease
single ventricle
Language English
License Copyright © 2017 American College of Cardiology Foundation. Published by Elsevier Inc. All rights reserved.
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PublicationTitle Journal of the American College of Cardiology
PublicationTitleAlternate J Am Coll Cardiol
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References 28571640 - J Am Coll Cardiol. 2017 Jun 6;69(22):2745-2747
29395194 - J Thorac Cardiovasc Surg. 2018 Apr;155(4):1727-1731
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Snippet Multicenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking. This study sought to describe...
Multicenter longitudinal objective data for survival into adulthood of patients who have undergone Fontan procedures are lacking.BACKGROUNDMulticenter...
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StartPage 2735
SubjectTerms Adolescent
Child
Cross-Sectional Studies
Echocardiography
Female
Follow-Up Studies
Fontan Procedure - methods
Forecasting
Health Status
Heart Defects, Congenital - diagnosis
Heart Defects, Congenital - mortality
Heart Defects, Congenital - surgery
Heart Ventricles - abnormalities
Heart Ventricles - diagnostic imaging
Humans
Male
Ontario - epidemiology
Quality of Life
Retrospective Studies
Risk Factors
Survival Rate - trends
Treatment Outcome
United States - epidemiology
Young Adult
Title Longitudinal Outcomes of Patients With Single Ventricle After the Fontan Procedure
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