H ferritin knockout mice: a model of hyperferritinemia in the absence of iron overload

Blood. 2001 Aug 1;98(3):525-32. doi: 10.1182/blood.v98.3.525.

Abstract

Ferritin, the iron-storing molecule, is made by the assembly of various proportions of 2 different H and L subunits into a 24-mer protein shell. These heteropolymers have distinct physicochemical properties, owing to the ferroxidase activity of the H subunit, which is necessary for iron uptake by the ferritin molecule, and the ability of the L subunit to facilitate iron core formation inside the protein shell. It has previously been shown that H ferritin is indispensable for normal development, since inactivation of the H ferritin gene by homologous recombination in mice is lethal at an early stage during embryonic development. Here the phenotypic analysis of the mice heterozygous for the H ferritin gene (Fth(+/-) mice) is reported, and differences in gene regulation between the 2 subunits are shown. The heterozygous Fth(+/-) mice were healthy and fertile and did not present any apparent abnormalities. Although they had iron-overloaded spleens at the adult stage, this is identical to what is observed in normal Fth(+/+) mice. However, these heterozygous mice had slightly elevated tissue L ferritin content and 7- to 10-fold more L ferritin in the serum than normal mice, but their serum iron remained unchanged. H ferritin synthesis from the remaining allele was not up-regulated. This probably results from subtle changes in the intracellular labile iron pool, which would stimulate L ferritin but not H ferritin synthesis. These results raise the possibility that reduced H ferritin expression might be responsible for unexplained human cases of hyperferritinemia in the absence of iron overload where the hereditary hyperferritinemia-cataract syndrome has been excluded. (Blood. 2001;98:525-532)

MeSH terms

  • Alleles
  • Anemia, Iron-Deficiency / genetics
  • Anemia, Iron-Deficiency / metabolism
  • Animals
  • Disease Models, Animal*
  • Ferritins / blood
  • Ferritins / deficiency*
  • Ferritins / genetics
  • Ferritins / metabolism
  • Homeostasis
  • Immunohistochemistry
  • Iron Overload / blood
  • Iron-Regulatory Proteins
  • Iron-Sulfur Proteins / metabolism
  • Mice
  • Mice, Knockout / genetics
  • Mice, Knockout / metabolism*
  • Protein Subunits
  • RNA-Binding Proteins / metabolism
  • Tissue Distribution

Substances

  • Iron-Regulatory Proteins
  • Iron-Sulfur Proteins
  • Protein Subunits
  • RNA-Binding Proteins
  • Ferritins