Liver Transplantation Trends and Outcomes for Hereditary Hemorrhagic Telangiectasia in the United States

Transplantation. 2019 Jul;103(7):1418-1424. doi: 10.1097/TP.0000000000002491.

Abstract

Background: Liver arteriovenous malformations (AVM) in hereditary hemorrhagic telangiectasia (HHT) can necessitate liver transplantation. There is limited data on HHT patients undergoing liver transplantation (LT) in the United States.

Methods: Two sources of data were used: (1) Scientific Registry of Transplant Recipients (SRTR) database (1998-2016) (2) Single center liver transplant database (Mayo Clinic Rochester, MN). The aims of this study were (1) to determine trends in LT for HHT-related liver involvement in the United States using the SRTR database; (2) to identify clinical characteristics, indications, and outcomes for LT in HHT.

Results: Thirty-nine HHT patients were listed for LT in the SRTR database from 1998-2016 to 1998-2001 (n = 1); 2002-2005 (n = 4); 2006-2010 (n = 10), and 2011-2016 (n = 24). Twenty-four underwent LT at a median age of 47.5 years (interquartile range, 37.0-58.5 years). Median calculated MELD score at time of LT was 8.0 (interquartile range, 7.0-9.5), and 75% received an exception MELD score. Two status-1 patients died during transplant surgery. Nineteen (86%) patients were alive after a median post-LT follow-up of 48 months, whereas 2 patients were lost to follow-up. Five of the aforementioned HHT patients underwent LT at Mayo Clinic, 4 with high output cardiac failure, and 1 with biliary ischemia. All 5 were alive at the time of last follow-up with good graft function and resolution of heart failure.

Conclusions: Outcomes after LT for HHT patients are excellent with 86% survival after a median follow-up of 48 months and resolution of heart failure. LT listing for HHT has increased in substantially in more recent eras.

MeSH terms

  • Adult
  • Aged
  • Cardiac Output, High / epidemiology
  • Cardiac Output, High / physiopathology
  • Databases, Factual
  • Female
  • Graft Survival
  • Heart Failure / epidemiology
  • Heart Failure / physiopathology
  • Humans
  • Liver Failure / diagnosis
  • Liver Failure / mortality
  • Liver Failure / physiopathology
  • Liver Failure / surgery*
  • Liver Transplantation / adverse effects
  • Liver Transplantation / mortality
  • Liver Transplantation / trends*
  • Male
  • Middle Aged
  • Outcome and Process Assessment, Health Care / trends*
  • Recovery of Function
  • Registries
  • Retrospective Studies
  • Telangiectasia, Hereditary Hemorrhagic / diagnosis
  • Telangiectasia, Hereditary Hemorrhagic / mortality
  • Telangiectasia, Hereditary Hemorrhagic / physiopathology
  • Telangiectasia, Hereditary Hemorrhagic / surgery*
  • Time Factors
  • Treatment Outcome
  • United States / epidemiology
  • Ventricular Function, Left