Milojevic, S., Ghosh, A., Makevic, V., Stojkovic, M., Capovilla, M.; Tosti, T., Budimirovic, D. B. & Protic, D. (2024). Circadian rhythm and sleep analyses in a fruit fly model of Fragile X Syndrome using a video-based automated behavioral research system. International Journal of Molecular Sciences, 25(14), 7949.
doi: 10.3390/ijms25147949
Abstract
Fragile X syndrome (FXS) is caused by the full mutation in the FMR1 gene on the Xq27.3 chromosome region. It is the most common monogenic cause of autism spectrum disorder (ASD) and inherited intellectual disability (ID). Besides ASD and ID and other symptoms, individuals with FXS may exhibit sleep problems and impairment of circadian rhythm (CR). The Drosophila melanogaster models of FXS, such as dFMR1B55, represent excellent models for research in the FXS field. During this study, sleep patterns and CR in dFMR1B55 mutants were analyzed, using a new platform based on continuous high-resolution videography integrated with a highly-customized version of an open-source software. This methodology provides more sensitive results, which could be crucial for all further research in this model of fruit flies. The study revealed that dFMR1B55 male mutants sleep more and can be considered weak rhythmic flies rather than totally arrhythmic and present a good alternative animal model of genetic disorder, which includes impairment of CR and sleep behavior. The combination of affordable videography and software used in the current study is a significant improvement over previous methods and will enable broader adaptation of such high-resolution behavior monitoring methods.
Keywords
Drosophila melanogaster model of fragile X syndrome; FMR1 gene; Fragile X syndrome; Circadian rhythm; Sleep
