Variants within the SP110 nuclear body protein modify risk of canine degenerative myelopathy

Proc Natl Acad Sci U S A. 2016 May 31;113(22):E3091-100. doi: 10.1073/pnas.1600084113. Epub 2016 May 16.

Abstract

Canine degenerative myelopathy (DM) is a naturally occurring neurodegenerative disease with similarities to some forms of amyotrophic lateral sclerosis (ALS). Most dogs that develop DM are homozygous for a common superoxide dismutase 1 gene (SOD1) mutation. However, not all dogs homozygous for this mutation develop disease. We performed a genome-wide association analysis in the Pembroke Welsh Corgi (PWC) breed comparing DM-affected and -unaffected dogs homozygous for the SOD1 mutation. The analysis revealed a modifier locus on canine chromosome 25. A haplotype within the SP110 nuclear body protein (SP110) was present in 40% of affected compared with 4% of unaffected dogs (P = 1.5 × 10(-5)), and was associated with increased probability of developing DM (P = 4.8 × 10(-6)) and earlier onset of disease (P = 1.7 × 10(-5)). SP110 is a nuclear body protein involved in the regulation of gene transcription. Our findings suggest that variations in SP110-mediated gene transcription may underlie, at least in part, the variability in risk for developing DM among PWCs that are homozygous for the disease-related SOD1 mutation. Further studies are warranted to clarify the effect of this modifier across dog breeds.

Keywords: ALS; SOD1; SP110; amyotrophic lateral sclerosis; degenerative myelopathy.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age of Onset
  • Animals
  • Disease Models, Animal
  • Dog Diseases / genetics*
  • Dog Diseases / pathology
  • Dogs
  • Female
  • Genome-Wide Association Study
  • Homozygote
  • Male
  • Muscular Diseases / genetics*
  • Muscular Diseases / pathology
  • Mutation / genetics*
  • Neurodegenerative Diseases / genetics*
  • Neurodegenerative Diseases / pathology
  • Nuclear Proteins / genetics*
  • RNA, Messenger / genetics
  • Real-Time Polymerase Chain Reaction
  • Reverse Transcriptase Polymerase Chain Reaction
  • Spinal Cord Diseases / genetics*
  • Spinal Cord Diseases / pathology
  • Superoxide Dismutase / genetics*

Substances

  • Nuclear Proteins
  • RNA, Messenger
  • Superoxide Dismutase

Associated data

  • GENBANK/KP245899
  • GENBANK/KP245902