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Conserved Cis-Acting Range Extender Element Mediates Extreme Long-Range Enhancer Activity in Mammals.
Bower, Grace; Hollingsworth, Ethan W; Jacinto, Sandra; Clock, Benjamin; Cao, Kaitlyn; Liu, Mandy; Dziulko, Adam; Alcaina-Caro, Ana; Xu, Qianlan; Skowronska-Krawczyk, Dorota; Lopez-Rios, Javier; Dickel, Diane E; Bardet, Anaïs F; Pennacchio, Len A; Visel, Axel; Kvon, Evgeny Z.
Afiliación
  • Bower G; Department of Developmental and Cell Biology, University of California, Irvine, CA 92967, USA.
  • Hollingsworth EW; Department of Developmental and Cell Biology, University of California, Irvine, CA 92967, USA.
  • Jacinto S; Medical Scientist Training Program, University of California, Irvine, CA 92967, USA.
  • Clock B; Department of Developmental and Cell Biology, University of California, Irvine, CA 92967, USA.
  • Cao K; Department of Developmental and Cell Biology, University of California, Irvine, CA 92967, USA.
  • Liu M; Department of Developmental and Cell Biology, University of California, Irvine, CA 92967, USA.
  • Dziulko A; Department of Developmental and Cell Biology, University of California, Irvine, CA 92967, USA.
  • Alcaina-Caro A; Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory, Berkeley, CA 94720, USA.
  • Xu Q; Centro Andaluz de Biología del Desarrollo (CABD), CSIC-Universidad Pablo de Olavide-Junta de Andalucía, Seville, 41013, Spain.
  • Skowronska-Krawczyk D; Department of Physiology and Biophysics, Department of Ophthalmology, Center for Translational Vision Research, School of Medicine, University of California, Irvine, CA, USA.
  • Lopez-Rios J; Department of Physiology and Biophysics, Department of Ophthalmology, Center for Translational Vision Research, School of Medicine, University of California, Irvine, CA, USA.
  • Dickel DE; Centro Andaluz de Biología del Desarrollo (CABD), CSIC-Universidad Pablo de Olavide-Junta de Andalucía, Seville, 41013, Spain.
  • Bardet AF; Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory, Berkeley, CA 94720, USA.
  • Pennacchio LA; Institut de Génétique et de Biologie Moléculaire et Cellulaire (IGBMC), Université de Strasbourg, CNRS UMR7104, INSERM U1258, 67400 Illkirch, France.
  • Visel A; Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory, Berkeley, CA 94720, USA.
  • Kvon EZ; U.S. Department of Energy Joint Genome Institute, Walnut Creek, CA 94598, USA.
bioRxiv ; 2024 May 26.
Article en En | MEDLINE | ID: mdl-38826394
ABSTRACT
While most mammalian enhancers regulate their cognate promoters over moderate distances of tens of kilobases (kb), some enhancers act over distances in the megabase range. The sequence features enabling such extreme-distance enhancer-promoter interactions remain elusive. Here, we used in vivo enhancer replacement experiments in mice to show that short- and medium-range enhancers cannot initiate gene expression at extreme-distance range. We uncover a novel conserved cis-acting element, Range EXtender (REX), that confers extreme-distance regulatory activity and is located next to a long-range enhancer of Sall1. The REX element itself has no endogenous enhancer activity. However, addition of the REX to other short- and mid-range enhancers substantially increases their genomic interaction range. In the most extreme example observed, addition of the REX increased the range of an enhancer by an order of magnitude, from its native 71kb to 840kb. The REX element contains highly conserved [C/T]AATTA homeodomain motifs. These motifs are enriched around long-range limb enhancers genome-wide, including the ZRS, a benchmark long-range limb enhancer of Shh. Mutating the [C/T]AATTA motifs within the ZRS does not affect its limb-specific enhancer activity at short range, but selectively abolishes its long-range activity, resulting in severe limb reduction in knock-in mice. In summary, we identify a sequence signature globally associated with long-range enhancer-promoter interactions and describe a prototypical REX element that is necessary and sufficient to confer extreme-distance gene activation by remote enhancers.

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: BioRxiv Año: 2024 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: BioRxiv Año: 2024 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos