title: Drosophila regulatory factor X is necessary for ciliated sensory neuron differentiation.
abstract: Ciliated neurons play an important role in sensory perception in many animals. Modified cilia at dendrite endings serve as sites of sensory signal capture and transduction. We describe Drosophila mutations that affect the transcription factor RFX and genetic rescue experiments that demonstrate its central role in sensory cilium differentiation. Rfx mutant flies show defects in chemosensory and mechanosensory behaviors but have normal phototaxis, consistent with Rfx expression in ciliated sensory neurons and neuronal precursors but not in photoreceptors. The mutant behavioral phenotypes are correlated with abnormal function and structure of neuronal cilia, as shown by the loss of sensory transduction and by defects in ciliary morphology and ultrastructure. These results identify Rfx as an essential regulator of ciliated sensory neuron differentiation in Drosophila.
authors.affiliation: Centre de Génétique Moléculaire et Cellulaire, CNRS UMR-5534, Université Claude Bernard Lyon-1, 69622 Villeurbanne, France.
authors.forename: Raphaelle
authors.initials: R
authors.lastname: Dubruille
authors.forename: Anne
authors.initials: A
authors.lastname: Laurençon
authors.forename: Camille
authors.initials: C
authors.lastname: Vandaele
authors.forename: Emiko
authors.initials: E
authors.lastname: Shishido
authors.forename: Madeleine
authors.initials: M
authors.lastname: Coulon-Bublex
authors.forename: Peter
authors.initials: P
authors.lastname: Swoboda
authors.forename: Pierre
authors.initials: P
authors.lastname: Couble
authors.forename: Maurice
authors.initials: M
authors.lastname: Kernan
authors.forename: Bénédicte
authors.initials: B
authors.lastname: Durand
mesh_terms: D000595:Amino Acid Sequence; D000818:Animals; D002454:Cell Differentiation / Q000502:physiology*; D002923:Cilia / Q000502:physiology*; D004268:DNA-Binding Proteins / Q000235:genetics / Q000378:metabolism*; D029721:Drosophila Proteins / Q000235:genetics / Q000378:metabolism; D004331:Drosophila melanogaster / Q000235:genetics / Q000502:physiology*; D004594:Electrophysiology; D005260:Female; D017344:Genes, Insect; D006801:Humans; D008297:Male; D008969:Molecular Sequence Data; D009024:Morphogenesis; D009154:Mutation; D009475:Neurons, Afferent / Q000502:physiology* / Q000648:ultrastructure; D010641:Phenotype; D011993:Recombinant Fusion Proteins / Q000235:genetics / Q000378:metabolism; D000071701:Regulatory Factor X Transcription Factors; D016415:Sequence Alignment; D014157:Transcription Factors / Q000235:genetics / Q000378:metabolism*
chemical_list: D004268:DNA-Binding Proteins; D029721:Drosophila Proteins; D011993:Recombinant Fusion Proteins; D000071701:Regulatory Factor X Transcription Factors; D014157:Transcription Factors