RAG2 mutants alter DSB repair pathway choice in vivo and illuminate the nature of 'alternative NHEJ'

Nucleic Acids Res. 2014 Jun;42(10):6352-64. doi: 10.1093/nar/gku295. Epub 2014 Apr 20.

Abstract

DNA double-stranded breaks (DSBs) can be repaired by several mechanisms, including classical NHEJ (c-NHEJ) and a poorly defined, error-prone process termed alternative NHEJ (a-NHEJ). How cells choose between these alternatives to join physiologic DSBs remains unknown. Here, we show that deletion of RAG2's C-terminus allows a-NHEJ to repair RAG-mediated DSBs in developing lymphocytes from both c-NHEJ-proficient and c-NHEJ-deficient mice, demonstrating that the V(D)J recombinase influences repair pathway choice in vivo. Analysis of V(D)J junctions revealed that, contrary to expectation, junctional characteristics alone do not reliably distinguish between a-NHEJ and c-NHEJ. These data suggest that a-NHEJ is not necessarily mutagenic, and may be more prevalent than previously appreciated. Whole genome sequencing of a lymphoma arising in a p53(-/-) mouse bearing a C-terminal RAG2 truncation reveals evidence of a-NHEJ and also of aberrant recognition of DNA sequences resembling RAG recognition sites.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Antigens, Nuclear / genetics
  • DNA Breaks, Double-Stranded*
  • DNA End-Joining Repair*
  • DNA-Binding Proteins / genetics*
  • Genes, p53
  • Ku Autoantigen
  • Lymphoma / genetics
  • Mice
  • Mice, Knockout
  • Receptors, Antigen, T-Cell / genetics
  • Sequence Deletion
  • Translocation, Genetic
  • V(D)J Recombination

Substances

  • Antigens, Nuclear
  • DNA-Binding Proteins
  • Rag2 protein, mouse
  • Receptors, Antigen, T-Cell
  • Xrcc6 protein, mouse
  • Ku Autoantigen