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• W2016442425 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 [1Warren S.T Nelson D Advances in molecular analysis of fragile X syndrome.JAMA. 1994; 271: 536-542Crossref PubMed Scopus (160) Google Scholar, 2Imbert G Feng Y Nelson D.L Warren S.T Mandel J.-L FMR1 and mutations in fragile X syndrome molecular biology, biochemistry, and genetics.in: Wells R.D Warren S.T Genetic Instabilities and Hereditary Neurological Diseases. Academic Press, San Diego1998: 27-53Google Scholar]. 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 [3Hagerman R Clinical and diagnostic aspects of fragile X syndrome.in: Wells R.D Warren S.T Genetic Instabilities and Hereditary Neurological Diseases. Academic Press, San Diego1998: 15-25Google Scholar, 4Gould E.L Loesch D.Z Martin M.J Hagerman R.J Armstrong S.M Huggins R.M Melatonin profiles and sleep characteristics in boys with fragile X syndrome a preliminary study.Am. J. Med. Genet. 2000; 95: 307-315Crossref PubMed Scopus (77) Google Scholar], we investigated whether a loss-of-function mutation of dfmr1 affect circadian behavior [5Dunlap J.C Molecular bases for circadian clocks.Cell. 1999; 96: 271-290Abstract Full Text Full Text PDF PubMed Scopus (2213) Google Scholar, 6Hall J.C Genetics of biological rhythms in Drosophila.Adv. Genet. 1998; 33: 135-184Crossref Scopus (59) Google Scholar, 7Scully A.L Kay S.A Time flies for Drosophila.Cell. 2000; 100: 297-300Abstract Full Text Full Text PDF Scopus (60) Google Scholar, 8Young M.W Life's 24-hour clock molecular control of circadian rhythms in animal cells.Trends Biochem. Sci. 2000; 25: 601-606Abstract Full Text Full Text PDF Scopus (81) Google Scholar]. 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." @default.
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• W2016442425 date "2002-08-01" @default.
• W2016442425 modified "2023-10-11" @default.
• W2016442425 title "A Role for the Drosophila Fragile X-Related Gene in Circadian Output" @default.
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• W2016442425 doi "https://doi.org/10.1016/s0960-9822(02)01036-9" @default.
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