NCOA4 Deficiency Impairs Systemic Iron Homeostasis

Cell Rep. 2016 Jan 26;14(3):411-421. doi: 10.1016/j.celrep.2015.12.065. Epub 2016 Jan 14.

Abstract

The cargo receptor NCOA4 mediates autophagic ferritin degradation. Here we show that NCOA4 deficiency in a knockout mouse model causes iron accumulation in the liver and spleen, increased levels of transferrin saturation, serum ferritin, and liver hepcidin, and decreased levels of duodenal ferroportin. Despite signs of iron overload, NCOA4-null mice had mild microcytic hypochromic anemia. Under an iron-deprived diet (2-3 mg/kg), mice failed to release iron from ferritin storage and developed severe microcytic hypochromic anemia and ineffective erythropoiesis associated with increased erythropoietin levels. When fed an iron-enriched diet (2 g/kg), mice died prematurely and showed signs of liver damage. Ferritin accumulated in primary embryonic fibroblasts from NCOA4-null mice consequent to impaired autophagic targeting. Adoptive expression of the NCOA4 COOH terminus (aa 239-614) restored this function. In conclusion, NCOA4 prevents iron accumulation and ensures efficient erythropoiesis, playing a central role in balancing iron levels in vivo.

Keywords: NCOA4; autophagy; ferritin; hypochromic anemia; iron.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Anemia, Hypochromic / metabolism
  • Anemia, Hypochromic / pathology
  • Animals
  • Autophagy / drug effects
  • Cell Line
  • Duodenum / metabolism
  • Duodenum / pathology
  • Erythrocytes / cytology
  • Erythrocytes / metabolism
  • Erythropoiesis / drug effects
  • Female
  • Ferritins / metabolism
  • Hepcidins / metabolism
  • Iron / metabolism*
  • Iron Overload / mortality
  • Iron Overload / pathology
  • Iron, Dietary / pharmacology
  • Liver / metabolism
  • Liver / pathology
  • Male
  • Mice
  • Mice, Knockout
  • Nuclear Receptor Coactivators / chemistry
  • Nuclear Receptor Coactivators / genetics
  • Nuclear Receptor Coactivators / metabolism*
  • Oxidoreductases / metabolism
  • Reactive Oxygen Species / metabolism
  • Spleen / metabolism
  • Spleen / pathology
  • Up-Regulation / drug effects

Substances

  • Hepcidins
  • Iron, Dietary
  • NcoA4 protein, mouse
  • Nuclear Receptor Coactivators
  • Reactive Oxygen Species
  • Ferritins
  • Iron
  • Oxidoreductases

Supplementary concepts

  • Anemia, hypochromic microcytic