Please use this identifier to cite or link to this item: https://biore.bio.bg.ac.rs/handle/123456789/5281
Title: 𝘋𝘳𝘰𝘴𝘰𝘱𝘩𝘪𝘭𝘢 𝘮𝘦𝘭𝘢𝘯𝘰𝘨𝘢𝘴𝘵𝘦𝘳 as a Model to Study Fragile X-Associated Disorders
Authors: Trajković, Jelena 
Makević, Vedrana
Pešić, Milica
Pavković-Lučić, Sofija 
Milojević, Sara
Cvjetković, Smiljana
Hagerman, Randi
Budimirović, Dejan
Protić, Dragana
Keywords: Fragile X syndrome;FXTAS;FMR1 gene;FMRP;Drosophila melanogaster
Issue Date: 2023
Rank: M22
Publisher: MDPI
Journal: Genes
Volume: 14
Issue: 1
Abstract: 
Fragile X syndrome (FXS) is a global neurodevelopmental disorder caused by the expansion of CGG trinucleotide repeats (≥200) in the Fragile X Messenger Ribonucleoprotein 1 (FMR1) gene. FXS is the hallmark of Fragile X-associated disorders (FXD) and the most common monogenic cause of inherited intellectual disability and autism spectrum disorder. There are several animal models used to study FXS. In the FXS model of Drosophila, the only ortholog of FMR1, dfmr1, is mutated so that its protein is missing. This model has several relevant phenotypes, including defects in the circadian output pathway, sleep problems, memory deficits in the conditioned courtship and olfactory conditioning paradigms, deficits in social interaction, and deficits in neuronal development. In addition to FXS, a model of another FXD, Fragile X-associated tremor/ataxia syndrome (FXTAS), has also been established in Drosophila. This review summarizes many years of research on FXD in Drosophila models.
URI: https://biore.bio.bg.ac.rs/handle/123456789/5281
ISSN: 2073-4425
DOI: 10.3390/genes14010087
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