Fontan-associated protein-losing enteropathy and post‒heart transplant outcomes: A multicenter study

J Heart Lung Transplant. 2019 Jan;38(1):17-25. doi: 10.1016/j.healun.2018.09.024. Epub 2018 Oct 2.

Abstract

Background: The influence of Fontan-associated protein-losing enteropathy's (PLE) severity, duration, and treatment on heart transplant (HTx) outcomes is unknown. We hypothesized that long-standing PLE and PLE requiring more intensive therapy are associated with increased post-HTx mortality.

Methods: This 12-center, retrospective cohort study of post-Fontan patients with PLE referred for HTx from 2003 to 2015 involved collection of demographic, medical, surgical, and catheterization data, as well as PLE-specific data, including duration of disease, intensity/details of treatment, hospitalizations, and complications. Factors associated with waitlist and post-HTx outcomes and PLE resolution were sought.

Results: Eighty patients (median of 5 per center) were referred for HTx evaluation. Of 68 patients listed for HTx, 8 were removed due to deterioration, 4 died waiting, and 4 remain listed. In 52 patients undergoing HTx, post-HTx 1-month survival was 92% and 1-year survival was 83%. PLE-specific factors, including duration of PLE pre-HTx, pre-HTx hospitalizations, need for/frequency of albumin replacement, PLE therapies, and growth parameters had no association with post-HTx mortality. Immunosuppressant regimen was associated with mortality; standard mycophenolate mofetil immunotherapy was used in 95% of survivors compared with only 44% of non-survivors (p = 0.03). Rejection (53%) and infection (42%) post-HTx were common, but not associated with PLE-specific factors. PLE resolved completely in all but 1 HTx survivor at a median of 1 month (interquartile range 1 to 3 months); resolution was not affected by PLE-specific factors.

Conclusions: PLE severity, duration, and treatment do not influence post-HTx outcome, but immunosuppressive regimen may have an impact on survival. PLE resolves in nearly all survivors.

Keywords: Fontan; congenital heart disease, protein-losing enteropathy; heart transplant; pediatric transplant; protein-losing enteropathy.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Female
  • Follow-Up Studies
  • Fontan Procedure / adverse effects*
  • Heart Defects, Congenital / surgery*
  • Heart Transplantation / adverse effects*
  • Humans
  • Infant
  • Male
  • Postoperative Complications*
  • Prognosis
  • Protein-Losing Enteropathies / epidemiology
  • Protein-Losing Enteropathies / etiology*
  • Retrospective Studies
  • Risk Factors
  • Survival Rate / trends
  • United States / epidemiology
  • Young Adult