title: A role for the Drosophila fragile X-related gene in circadian output.
abstract: Mutations that abolish expression of an X-linked gene, FMR1, result in the pathogenesis of fragile X syndrome, the most common form of inherited mental retardation. To understand the normal function of the FMR1 protein, we have produced fly strains bearing deletions in a Drosophila homolog of FMR1 (dfmr1). Since fragile X patients show a number of abnormal behaviors including sleep problems, we investigated whether a loss-of-function mutation of dfmr1 affect circadian behavior. Here we show that under constant darkness (DD), a lack of dfmr1 expression causes arrhythmic locomotor activity, but in light:dark cycles, their behavioral rhythms appear normal. In addition, the clock-controlled eclosion rhythm is normal in DFMR1-deficient flies. These results suggest that DFMR1 plays a critical role in the circadian output pathway regulating locomotor activity in Drosophila.
authors.affiliation: Institute for Genome Research, University of Tokushima, Kuramoto 3-18-15, 770-8503, Tokushima, Japan.
authors.forename: Shunsuke
authors.initials: S
authors.lastname: Inoue
authors.forename: Masami
authors.initials: M
authors.lastname: Shimoda
authors.forename: Izumi
authors.initials: I
authors.lastname: Nishinokubi
authors.forename: Mikiko C
authors.initials: MC
authors.lastname: Siomi
authors.forename: Miwako
authors.initials: M
authors.lastname: Okamura
authors.forename: Akira
authors.initials: A
authors.lastname: Nakamura
authors.forename: Satoru
authors.initials: S
authors.lastname: Kobayashi
authors.forename: Norio
authors.initials: N
authors.lastname: Ishida
authors.forename: Haruhiko
authors.initials: H
authors.lastname: Siomi
mesh_terms: D000818:Animals; D002940:Circadian Rhythm / Q000235:genetics*; D004330:Drosophila / Q000235:genetics / Q000502:physiology*; D029721:Drosophila Proteins; D051860:Fragile X Messenger Ribonucleoprotein 1; D005600:Fragile X Syndrome / Q000235:genetics*; D017353:Gene Deletion; D006801:Humans; D023421:Models, Animal; D016601:RNA-Binding Proteins / Q000235:genetics*; D013997:Time Factors; D014960:X Chromosome
chemical_list: D029721:Drosophila Proteins; D051860:Fragile X Messenger Ribonucleoprotein 1; D016601:RNA-Binding Proteins; C467328:FMR1 protein, Drosophila