Abstract
Fragile X syndrome is caused by the loss of the FMR1 gene product, fragile X mental retardation protein (FMRP). The loss of FMRP leads to altered circadian rhythm behaviors in both mouse and Drosophila; however, the molecular mechanism behind this phenomenon remains elusive. Here we performed a series of gene expression analyses, including of both mRNAs and microRNAs (miRNAs), and identified a number of mRNAs and miRNAs (miRNA-1 and miRNA-281) with circadian rhythm-dependent altered expression in dfmr1 mutant flies. Identification of these RNAs lays the foundation for future investigations of the molecular pathway(s) underlying the altered circadian rhythms associated with loss of dFmr1.
Highlights
Fragile X syndrome (FXS), one of the most common forms of inherited mental retardation, is characterized by mental retardation of variable severity, autistic behavior, macroorchidism in adult males, characteristic facial features, and hyperextensible joints [1]
FXS is mainly caused by a massive CGG trinucleotide repeat expansion within the 59 untranslated region (UTR) of the fragile X mental retardation 1 gene (FMR1), which results in abnormal DNA methylation of both a nearby CpG island and the repeat itself; as a result, the transcription of FMR1 is silenced [2,3,4,5,6]
We performed a series of gene expression analyses, including of both mRNAs and microRNAs, and identified a number of mRNAs and miRNAs with circadian rhythmdependent altered expression in dfmr1 mutant flies
Summary
Fragile X syndrome (FXS), one of the most common forms of inherited mental retardation, is characterized by mental retardation of variable severity, autistic behavior, macroorchidism in adult males, characteristic facial features, and hyperextensible joints [1]. FMRP, along with its autosomal paralogs, the fragile X-related proteins FXR1P and FXR2P, constitute a wellconserved, small family of RNA-binding proteins (the fragile Xrelated gene family) that share over 60% amino acid identity and contain two types of RNA-binding motifs: two ribonucleoprotein K-homology domains (KH domains) and a cluster of arginine and glycine residues (RGG box) [10,11]. Unlike their mammalian counterparts, the fly genome harbors a single Fmr gene homolog, referred to as dFmr. FMRP is found to form a messenger ribonucleoprotein (mRNP) complex that associates with translating polyribosomes [14]
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