Loss of DJ-1 elicits retinal abnormalities, visual dysfunction, and increased oxidative stress in mice

Exp Eye Res. 2015 Oct:139:22-36. doi: 10.1016/j.exer.2015.07.014. Epub 2015 Jul 26.

Abstract

DJ-1/PARK7 mutations or deletions cause autosomal recessive early onset Parkinson's disease (PD). Thus, DJ-1 protein has been extensively studied in brain and neurons. PD patients display visual symptoms; however, the visual symptoms specifically attributed to PD patients carrying DJ-1/PARK7 mutations are not known. In this study, we analyzed the structure and physiology of retinas of 3- and 6-month-old DJ-1 knockout (KO) mice to determine how loss of function of DJ-1 specifically contributes to the phenotypes observed in PD patients. As compared to controls, the DJ-1 KO mice displayed an increase in the amplitude of the scotopic ERG b-wave and cone ERG, while the amplitude of a subset of the dc-ERG components was decreased. The main structural changes in the DJ-1 KO retinas were found in the outer plexiform layer (OPL), photoreceptors and retinal pigment epithelium (RPE), which were observed at 3 months and progressively increased at 6 months. RPE thinning and structural changes within the OPL were observed in the retinas in DJ-1 KO mice. DJ-1 KO retinas also exhibited disorganized outer segments, central decrease in red/green cone opsin staining, decreased labeling of ezrin, broader distribution of ribeye labeling, decreased tyrosine hydroxylase in dopaminergic neurons, and increased 7,8-dihydro-8-oxoguanine-labeled DNA oxidation. Accelerated outer retinal atrophy was observed in DJ-1 KO mice after selective oxidative damage induced by a single tail vein injection of NaIO3, exposing increased susceptibility to oxidative stress. Our data indicate that DJ-1-deficient retinas exhibit signs of morphological abnormalities and physiological dysfunction in association with increased oxidative stress. Degeneration of RPE cells in association with oxidative stress is a key hallmark of age-related macular degeneration (AMD). Therefore, in addition to detailing the visual defects that occur as a result of the absence of DJ-1, our data is also relevant to AMD pathogenesis.

Keywords: Biochemistry; DJ-1 knockout; Histology; Immunohistology; Morphology; Oxidation; Physiology; Retina.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural
  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, Non-P.H.S.

MeSH terms

  • Animals
  • Blotting, Western
  • DNA / genetics*
  • DNA Mutational Analysis
  • Disease Models, Animal
  • Electroretinography
  • Female
  • Genotype
  • Immunohistochemistry
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Microscopy, Electron, Transmission
  • Mutation*
  • Oncogene Proteins / biosynthesis
  • Oncogene Proteins / genetics*
  • Oxidative Stress
  • Peroxiredoxins / biosynthesis
  • Peroxiredoxins / genetics*
  • Polymerase Chain Reaction
  • Protein Deglycase DJ-1
  • Retinal Degeneration / genetics*
  • Retinal Degeneration / pathology
  • Retinal Degeneration / physiopathology
  • Retinal Pigment Epithelium / metabolism*
  • Retinal Pigment Epithelium / physiopathology
  • Retinal Pigment Epithelium / ultrastructure
  • Signal Transduction

Substances

  • Oncogene Proteins
  • DNA
  • Peroxiredoxins
  • PARK7 protein, mouse
  • Protein Deglycase DJ-1