Defective Hfp-dependent transcriptional repression of dMYC is fundamental to tissue overgrowth in Drosophila XPB models

Nat Commun. 2015 Jun 15:6:7404. doi: 10.1038/ncomms8404.

Abstract

Nucleotide excision DNA repair (NER) pathway mutations cause neurodegenerative and progeroid disorders (xeroderma pigmentosum (XP), Cockayne syndrome (CS) and trichothiodystrophy (TTD)), which are inexplicably associated with (XP) or without (CS/TTD) cancer. Moreover, cancer progression occurs in certain patients, but not others, with similar C-terminal mutations in the XPB helicase subunit of transcription and NER factor TFIIH. Mechanisms driving overproliferation and, therefore, cancer associated with XPB mutations are currently unknown. Here using Drosophila models, we provide evidence that C-terminally truncated Hay/XPB alleles enhance overgrowth dependent on reduced abundance of RNA recognition motif protein Hfp/FIR, which transcriptionally represses the MYC oncogene homologue, dMYC. The data demonstrate that dMYC repression and dMYC-dependent overgrowth in the Hfp hypomorph is further impaired in the C-terminal Hay/XPB mutant background. Thus, we predict defective transcriptional repression of MYC by the Hfp orthologue, FIR, might provide one mechanism for cancer progression in XP/CS.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cell Proliferation / genetics*
  • Chromatin Immunoprecipitation
  • DNA Helicases / genetics
  • DNA-Binding Proteins / genetics*
  • Drosophila Proteins / genetics*
  • Drosophila melanogaster
  • Gene Expression Regulation
  • Guanine Nucleotide Exchange Factors / genetics*
  • Immunohistochemistry
  • Mutation
  • Transcription Factors / genetics*
  • Transcription, Genetic
  • Xeroderma Pigmentosum / genetics

Substances

  • DNA-Binding Proteins
  • Drosophila Proteins
  • Guanine Nucleotide Exchange Factors
  • Hfp protein, Drosophila
  • Myc protein, Drosophila
  • Transcription Factors
  • hay protein, Drosophila
  • DNA Helicases