Gigaxonin mutation analysis in patients with NIFID

Neurobiol Aging. 2011 Aug;32(8):1528-9. doi: 10.1016/j.neurobiolaging.2009.08.018. Epub 2009 Sep 26.

Abstract

Neuronal intermediate filament inclusion disease (NIFID) is a frontotemporal lobar degeneration (FTLD) characterized by frontotemporal dementia (FTD), pyramidal and extrapyramidal signs. The disease is histologically characterized by the presence of abnormal neuronal cytoplasmic inclusions (NCIs) which contain α-internexin and other neuronal intermediate filament (IF) proteins. Gigaxonin (GAN) is a cytoskeletal regulating protein and the genetic cause of giant axonal neuropathy. Since the immunoreactive profile of NCIs in NIFID is similar to that observed in brain sections from Gan(Δex1/Δex1) mice, we speculated that GAN could be a candidate gene causing NIFID. Therefore, we performed a mutation analysis of GAN in NIFID patients. Although the NCIs of NIFID and Gan(Δex1/Δex1) mice were immunohistochemically similar, no GAN variant was identified in DNA obtained from well-characterized cases of NIFID.

MeSH terms

  • Animals
  • Cytoskeletal Proteins / genetics*
  • Cytoskeletal Proteins / metabolism
  • Disease Models, Animal*
  • Frontotemporal Lobar Degeneration / genetics*
  • Frontotemporal Lobar Degeneration / metabolism
  • Frontotemporal Lobar Degeneration / pathology
  • Humans
  • Intermediate Filaments / metabolism
  • Intermediate Filaments / pathology
  • Mice
  • Mice, Mutant Strains
  • Phenotype

Substances

  • Cytoskeletal Proteins
  • GAN protein, human
  • Gan protein, mouse