An Overview of the Ferroptosis Hallmarks in Friedreich's Ataxia

Biomolecules. 2020 Oct 28;10(11):1489. doi: 10.3390/biom10111489.

Abstract

Background: Friedreich's ataxia (FRDA) is a neurodegenerative disease characterized by early mortality due to hypertrophic cardiomyopathy. FRDA is caused by reduced levels of frataxin (FXN), a mitochondrial protein involved in the synthesis of iron-sulphur clusters, leading to iron accumulation at the mitochondrial level, uncontrolled production of reactive oxygen species and lipid peroxidation. These features are also common to ferroptosis, an iron-mediated type of cell death triggered by accumulation of lipoperoxides with distinct morphological and molecular characteristics with respect to other known cell deaths.

Scope of review: Even though ferroptosis has been associated with various neurodegenerative diseases including FRDA, the mechanisms leading to disease onset/progression have not been demonstrated yet. We describe the molecular alterations occurring in FRDA that overlap with those characterizing ferroptosis.

Major conclusions: The study of ferroptotic pathways is necessary for the understanding of FRDA pathogenesis, and anti-ferroptotic drugs could be envisaged as therapeutic strategies to cure FRDA.

Keywords: FRDA; GPX4; Iron metabolism; ROS; frataxin; lipid metabolism; neurodegeneration.

Publication types

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

MeSH terms

  • Cardiomyopathy, Hypertrophic
  • Ferroptosis / genetics*
  • Frataxin
  • Friedreich Ataxia / epidemiology*
  • Friedreich Ataxia / genetics
  • Friedreich Ataxia / pathology
  • Humans
  • Iron / metabolism
  • Iron-Binding Proteins / genetics*
  • Lipid Peroxidation / genetics
  • Mitochondria / genetics
  • Mitochondria / pathology
  • Neurodegenerative Diseases / epidemiology*
  • Neurodegenerative Diseases / genetics
  • Neurodegenerative Diseases / parasitology
  • Oxidative Stress / genetics
  • Reactive Oxygen Species / metabolism

Substances

  • Iron-Binding Proteins
  • Reactive Oxygen Species
  • Iron