A requirement of FancL and FancD2 monoubiquitination in DNA repair

Sohsuke Seki, Mioko Ohzeki, Akiko Uchida, Seiki Hirano, Nobuko Matsushita, Hiroyuki Kitao, Tsukasa Oda, Takayuki Yamashita, Naoki Kashihara, Akio Tsubahara, Minoru Takata, Masamichi Ishiai

Research output: Contribution to journalArticlepeer-review

32 Citations (Scopus)


The rare hereditary disorder Fanconi anemia (FA) can be caused by mutations in components of the FA core complex (FancA/B/C/E/F/G/L/M), a key regulator FancD2, the breast cancer susceptibility protein BRCA2/ FancD1, or the newly identified FancJ/BRIP1 helicase. By performing yeast two-hybrid (Y2H) screens using N-terminal chicken (ch) FancD2 as a bait, we have identified chFancL, the likely ubiquitin E3 ligase subunit of the FA core complex. We also found that ectopically expressed FancD2 and FancL co-immunoprecipitated in 293T cells, and this interaction was dependent on the PHD domain of FancL. FANCL-disrupted chicken DT40 cells displayed defects in both FancD2 monoubiquitination and focus formation. Importantly, cell lines lacking the FANCL or FANCD2 genes, or carrying a "knock-in" mutation of the FancD2 monoubiquitination site (where the Lys 563 residue is changed to Arg), displayed quantitatively identical defects in the repair of I-SceI-induced chromosomal breaks by homologous recombination (HR). These data establish the role of FANCL and FancD2 monoubiquitination in HR repair.

Original languageEnglish
Pages (from-to)299-310
Number of pages12
JournalGenes to Cells
Issue number3
Publication statusPublished - Mar 2007
Externally publishedYes

All Science Journal Classification (ASJC) codes

  • Genetics
  • Cell Biology


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