DNA-PKcs mutations in dogs and horses: allele frequency and association with neoplasia

Gene. 2002 Jan 23;283(1-2):263-9. doi: 10.1016/s0378-1119(01)00880-0.

Abstract

Previously, spontaneous genetic immunodeficiencies in mice, Arabian foals, and recently in Jack Russell terriers have been ascribed to defects in DNA-PKcs (catalytic subunit of the DNA dependent protein kinase) expression. In severe combined immunodeficiency (SCID) foals, a 5 bp deletion at codon 9480 results in a frameshift and a 967 amino acid deletion from the C terminus (including the entire PI3 kinase domain) and an unstable mutant protein. In SCID mice, a single base pair mutation results in a premature stop codon and deletion of 83 amino acids; as in SCID foals, the mutant protein is unstable. Here, we define the mutation within the canine DNA-PKcs gene that results in SCID. In this case, a point mutation results in a stop codon at nucleotide 10,828 and premature termination at a position 517 amino acids before the normal C terminus resulting in a functionally null allele. Thus, this is the third documentation of a spontaneous germline mutation in the C terminus of DNA-PKcs. Emerging data implicate DNA repair factors as potential tumor suppressors. Here, we have ascertained the carrier frequency of the defective DNA-PKcs genes in Arabian horses and in Jack Russell terriers. Our data indicate (in good agreement with a previous report) that the carrier frequency of the equine SCID allele is approximately 8%; in contrast, the carrier frequency of the canine SCID allele is less than 1.1%. We also assessed the frequency of the equine SCID allele in a series of 295 tumors from Arabian horses. We find a statistically significant correlation between the development of a virally induced tumor (sarcoid) and heterozygosity for the equine SCID allele. These data provide further support for an emerging consensus: that DNA-PK may normally act as a tumor suppressor through its caretaker role in maintaining chromosomal stability.

Publication types

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

MeSH terms

  • Amino Acid Sequence
  • Animals
  • Base Sequence
  • Catalytic Domain / genetics*
  • Codon, Nonsense
  • DNA Mutational Analysis
  • DNA, Complementary / chemistry
  • DNA, Complementary / genetics
  • DNA, Neoplasm / genetics
  • DNA-Activated Protein Kinase
  • DNA-Binding Proteins*
  • Dogs
  • Gene Frequency
  • Genotype
  • Heterozygote
  • Horses
  • Molecular Sequence Data
  • Mutation
  • Neoplasms / enzymology
  • Neoplasms / genetics
  • Neoplasms / pathology
  • Point Mutation
  • Protein Serine-Threonine Kinases / genetics*

Substances

  • Codon, Nonsense
  • DNA, Complementary
  • DNA, Neoplasm
  • DNA-Binding Proteins
  • DNA-Activated Protein Kinase
  • Protein Serine-Threonine Kinases

Associated data

  • GENBANK/AF448228