Absence of mitochondrial superoxide dismutase results in a murine hemolytic anemia responsive to therapy with a catalytic antioxidant

J Exp Med. 2001 Apr 16;193(8):925-34. doi: 10.1084/jem.193.8.925.

Abstract

Manganese superoxide dismutase 2 (SOD2) is a critical component of the mitochondrial pathway for detoxification of O2(-), and targeted disruption of this locus leads to embryonic or neonatal lethality in mice. To follow the effects of SOD2 deficiency in cells over a longer time course, we created hematopoietic chimeras in which all blood cells are derived from fetal liver stem cells of Sod2 knockout, heterozygous, or wild-type littermates. Stem cells of each genotype efficiently rescued hematopoiesis and allowed long-term survival of lethally irradiated host animals. Peripheral blood analysis of leukocyte populations revealed no differences in reconstitution kinetics of T cells, B cells, or myeloid cells when comparing Sod2(+/+), Sod2(-/-), and Sod2(+/-) fetal liver recipients. However, animals receiving Sod2(-/-) cells were persistently anemic, with findings suggestive of a hemolytic process. Loss of SOD2 in erythroid progenitor cells results in enhanced protein oxidative damage, altered membrane deformation, and reduced survival of red cells. Treatment of anemic animals with Euk-8, a catalytic antioxidant with both SOD and catalase activities, significantly corrected this oxidative stress-induced condition. Such therapy may prove useful in treatment of human disorders such as sideroblastic anemia, which SOD2 deficiency most closely resembles.

Publication types

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

MeSH terms

  • Anemia, Hemolytic / blood
  • Anemia, Hemolytic / drug therapy*
  • Anemia, Hemolytic / genetics*
  • Animals
  • Antioxidants / therapeutic use*
  • B-Lymphocytes / immunology
  • Bone Marrow Cells / immunology
  • Cell Transplantation
  • Ethylenediamines / therapeutic use*
  • Fetal Tissue Transplantation / physiology
  • Free Radical Scavengers / therapeutic use
  • Genotype
  • Heterozygote
  • Isoenzymes / deficiency
  • Isoenzymes / genetics
  • Liver / cytology
  • Mice
  • Mice, Knockout
  • Mitochondria / enzymology*
  • Organometallic Compounds / therapeutic use*
  • Polymerase Chain Reaction
  • Spleen / immunology
  • Superoxide Dismutase / deficiency*
  • Superoxide Dismutase / genetics
  • T-Lymphocytes / immunology
  • Thymus Gland / immunology
  • Transplantation Chimera

Substances

  • Antioxidants
  • Ethylenediamines
  • Free Radical Scavengers
  • Isoenzymes
  • Organometallic Compounds
  • N,N'-bis(salicylideneamino)ethane-manganese(II)
  • Superoxide Dismutase