Mechanistic insight into site-restricted monoubiquitination of FANCD2 by Ube2t, FANCL, and FANCI

Mol Cell. 2008 Dec 26;32(6):767-77. doi: 10.1016/j.molcel.2008.12.003.

Abstract

A key step in the Fanconi anemia (FA) tumor suppressor pathway is the site-specific monoubiquitination of the FANCD2 protein. Genetic studies indicate that this crucial modification requires eight known FA gene products and the E2-conjugating enzyme Ube2t. Here, we minimally reconstitute this monoubiquitination reaction with Ube2t and the FANCL protein, revealing that monoubiquitination is stimulated by a conserved RWD-like domain in FANCL. Furthermore, addition of the FANCI protein enhances monoubiquitination and also restricts it to the in vivo substrate lysine residue on FANCD2. This work therefore establishes a system that provides mechanistic insight into the functions of FANCL and FANCI in the catalysis of FANCD2 monoubiquitination.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amino Acid Sequence
  • Animals
  • Cell Line
  • Chickens
  • Conserved Sequence
  • Fanconi Anemia Complementation Group D2 Protein / metabolism*
  • Fanconi Anemia Complementation Group L Protein / chemistry
  • Fanconi Anemia Complementation Group L Protein / metabolism*
  • Fanconi Anemia Complementation Group Proteins / metabolism*
  • Humans
  • Molecular Sequence Data
  • Protein Structure, Tertiary
  • Substrate Specificity
  • Ubiquitin-Conjugating Enzymes / metabolism*
  • Ubiquitination*

Substances

  • FANCI protein, human
  • Fanconi Anemia Complementation Group D2 Protein
  • Fanconi Anemia Complementation Group Proteins
  • UBE2T protein, human
  • UBE2W protein, human
  • Ubiquitin-Conjugating Enzymes
  • Fanconi Anemia Complementation Group L Protein