Autoubiquitylation of the V(D)J recombinase protein RAG1

Proc Natl Acad Sci U S A. 2003 Dec 23;100(26):15446-51. doi: 10.1073/pnas.2637012100. Epub 2003 Dec 11.

Abstract

V(D)J recombination, the rearrangement of gene segments to assemble Ig and T cell receptor coding regions, is vital to B and T lymphocyte development. Here, we demonstrate that the V(D)J recombinase protein RAG1 undergoes ubiquitylation in cells. In vitro, the RING finger domain of RAG1 acts as a ubiquitin ligase that mediates its own ubiquitylation at a highly conserved K residue in the RAG1 amino-terminal region. Ubiquitylation is best supported by a specific ubiquitin-conjugating enzyme, UbcH3/CDC34, and requires an intact RAG1 RING finger motif. Disruption of the RING finger and certain RAG1 N-terminal truncations are associated with immunodeficiency in human patients, suggesting that RAG1's ubiquitin ligase is required for its biological role in lymphocyte development.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Base Sequence
  • Binding Sites
  • Cloning, Molecular
  • DNA Primers
  • DNA-Binding Proteins / chemistry
  • DNA-Binding Proteins / metabolism
  • Homeodomain Proteins / chemistry
  • Homeodomain Proteins / genetics
  • Homeodomain Proteins / metabolism*
  • Humans
  • Kinetics
  • Mice
  • Nuclear Proteins
  • Polymerase Chain Reaction
  • Recombinant Proteins / chemistry
  • Recombinant Proteins / metabolism
  • Sequence Alignment
  • Sequence Homology, Amino Acid
  • Ubiquitin / metabolism*
  • VDJ Recombinases / metabolism*
  • Zinc / metabolism

Substances

  • DNA Primers
  • DNA-Binding Proteins
  • Homeodomain Proteins
  • Nuclear Proteins
  • RAG2 protein, human
  • Rag2 protein, mouse
  • Recombinant Proteins
  • Ubiquitin
  • V(D)J recombination activating protein 2
  • RAG-1 protein
  • VDJ Recombinases
  • Zinc