Outcomes after liver transplantation in MPV17 deficiency (Navajo neurohepatopathy): A single-center case series

Pediatr Transplant. 2022 Aug;26(5):e14274. doi: 10.1111/petr.14274. Epub 2022 Apr 24.

Abstract

Background: MPV17-related mitochondrial DNA maintenance defect (MPV17 deficiency) is a rare, autosomal recessive mitochondrial DNA depletion syndrome with a high mortality rate in infancy and early childhood due to progression to liver failure. Liver transplantation for children with MPV17 deficiency has been considered controversial due to uncertainty about the potential progression of extrahepatic manifestations following liver transplantation.

Methods: We describe our institution's experience for two infants diagnosed with infantile MPV17 deficiency who presented in acute on chronic liver failure, but with normal development and normal neurological status who successfully underwent liver transplantation.

Results: Both patients underwent successful liver transplantation with normal development and neurological status at 3 years and 16 months post-transplant, respectively.

Conclusions: In this rare disease population, we describe two infants with MPV17 deficiency who underwent liver transplantation for acute on chronic liver failure who continue to have normal development, without progression of neurological disease. MPV17 deficiency should not be considered a contraindication to liver transplantation.

Keywords: MPV17; MPV17 deficiency; Navajo; acute liver failure; development; hepatocerebral; infantile; liver; mitochondrial; neurohepatopathy; neurological progression; pediatric liver transplant.

Publication types

  • Case Reports

MeSH terms

  • Acute-On-Chronic Liver Failure*
  • Child
  • Child, Preschool
  • DNA, Mitochondrial / genetics
  • Heredodegenerative Disorders, Nervous System
  • Humans
  • Infant
  • Liver Diseases
  • Liver Transplantation*
  • Membrane Proteins / genetics
  • Mitochondrial Diseases
  • Mitochondrial Proteins / genetics
  • Peripheral Nervous System Diseases

Substances

  • DNA, Mitochondrial
  • MPV17 protein, human
  • Membrane Proteins
  • Mitochondrial Proteins

Supplementary concepts

  • Navajo neurohepatopathy