title: XNP-1/ATR-X acts with RB, HP1 and the NuRD complex during larval development in C. elegans.
abstract: Mutations in the XNP/ATR-X gene cause several X-linked mental retardation syndromes in humans. The XNP/ATR-X gene encodes a DNA-helicase belonging to the SNF2 family. It has been proposed that XNP/ATR-X might be involved in chromatin remodelling. The lack of a mouse model for the ATR-X syndrome has, however, hampered functional studies of XNP/ATR-X. C. elegans possesses one homolog of the XNP/ATR-X gene, named xnp-1. By analysing a deletion mutant, we show that xnp-1 is required for the development of the embryo and the somatic gonad. Moreover, we show that abrogation of xnp-1 function in combination with inactivation of genes of the NuRD complex, as well as lin-35/Rb and hpl-2/HP1 leads to a stereotyped block of larval development with a cessation of growth but not of cell division. We also demonstrate a specific function for xnp-1 together with lin-35 or hpl-2 in the control of transgene expression, a process known to be dependent on chromatin remodelling. This study thus demonstrates that in vivo XNP-1 acts in association with RB, HP1 and the NuRD complex during development.
authors.affiliation: INSERM U491, Faculté de Médecine la Timone, 27, Bd Jean Moulin, 13385 Marseille Cedex 5, France.
authors.forename: Carlos
authors.initials: C
authors.lastname: Cardoso
authors.forename: Carole
authors.initials: C
authors.lastname: Couillault
authors.forename: Cecile
authors.initials: C
authors.lastname: Mignon-Ravix
authors.forename: Anne
authors.initials: A
authors.lastname: Millet
authors.forename: Jonathan J
authors.initials: JJ
authors.lastname: Ewbank
authors.forename: Michel
authors.initials: M
authors.lastname: Fontés
authors.forename: Nathalie
authors.initials: N
authors.lastname: Pujol
mesh_terms: D000818:Animals; D030801:Animals, Genetically Modified; D017173:Caenorhabditis elegans / Q000235:genetics / Q000254:growth & development* / Q000502:physiology*; D029742:Caenorhabditis elegans Proteins / Q000235:genetics / Q000502:physiology; D004265:DNA Helicases / Q000235:genetics / Q000502:physiology; D004268:DNA-Binding Proteins / Q000235:genetics / Q000502:physiology; D017238:Genes, Helminth; D006066:Gonads / Q000254:growth & development; D015801:Helminth Proteins / Q000235:genetics / Q000502:physiology*; D051537:Hepatocyte Nuclear Factor 1; D051538:Hepatocyte Nuclear Factor 1-alpha; D006655:Histone Deacetylases / Q000235:genetics / Q000502:physiology; D006801:Humans; D007814:Larva / Q000254:growth & development; D056245:Mi-2 Nucleosome Remodeling and Deacetylase Complex; D009154:Mutation; D009687:Nuclear Proteins / Q000235:genetics / Q000502:physiology; D034622:RNA Interference; D016160:Retinoblastoma Protein / Q000235:genetics / Q000502:physiology; D014157:Transcription Factors / Q000235:genetics / Q000502:physiology
chemical_list: D029742:Caenorhabditis elegans Proteins; D004268:DNA-Binding Proteins; C496487:HNF1A protein, human; D015801:Helminth Proteins; D051538:Hepatocyte Nuclear Factor 1-alpha; D009687:Nuclear Proteins; D016160:Retinoblastoma Protein; D014157:Transcription Factors; D051537:Hepatocyte Nuclear Factor 1; D006655:Histone Deacetylases; D056245:Mi-2 Nucleosome Remodeling and Deacetylase Complex; D004265:DNA Helicases; C121256:XNP-1 protein, C elegans