4.1 Article

Circadian Phenotypes of Drosophila Fragile X Mutants in Alternative Genetic Backgrounds

期刊

ZOOLOGICAL SCIENCE
卷 25, 期 6, 页码 561-571

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ZOOLOGICAL SOC JAPAN
DOI: 10.2108/zsj.25.561

关键词

fragile-X syndrome; behavior; locomotor rhythm; eclosion; genetic background; lateral neuron; luciferase assay

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资金

  1. JSPS [18570076]
  2. Grants-in-Aid for Scientific Research [18570076] Funding Source: KAKEN

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Drosophila FMR1 mutants are models of human fragile X syndrome. They show a loss of locomotor activity rhythm and severe degradation of eclosion timing. We analyzed the circadian behavior of FMR1 mutants (dfmr1(B55)) in two genetic backgrounds, yellow white (yw) and Canton S (CS). The arrhythmic phenotype of circadian locomotor activity in constant darkness (DD) did not significantly change in either genetic background. Surprisingly, eclosion timing was completely restored by backcrossing dfmr1(B55) with yw or CS flies. Morphological analysis of the small ventrally located lateral neurons of FMR1 mutants revealed that the dorsal-projection area was significantly larger in arrhythmic than rhythmic flies. In addition, dfmr1(B55) mutants in both genetic backgrounds had a significantly lower evening peak in the light-dark (LD) cycle. These results indicate that lack of FMR1 does not affect eclosion timing, but alters locomotor activity patterns in both LD and DD conditions by affecting the arborization of small ventrally located lateral neurons. Thus, the FMR1 gene may regulate the circadian-related locomotor activity of Drosophila.

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