Milojevic, S.; Ghosh, A.; Makevic, V.; Stojkovic, M.; Capovilla, M.; Tosti, T.; Budimirovic, D.; Protic, D. Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System. Int. J. Mol. Sci.2024, 25, 7949.
Milojevic, S.; Ghosh, A.; Makevic, V.; Stojkovic, M.; Capovilla, M.; Tosti, T.; Budimirovic, D.; Protic, D. Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System. Int. J. Mol. Sci. 2024, 25, 7949.
Milojevic, S.; Ghosh, A.; Makevic, V.; Stojkovic, M.; Capovilla, M.; Tosti, T.; Budimirovic, D.; Protic, D. Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System. Int. J. Mol. Sci.2024, 25, 7949.
Milojevic, S.; Ghosh, A.; Makevic, V.; Stojkovic, M.; Capovilla, M.; Tosti, T.; Budimirovic, D.; Protic, D. Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System. Int. J. Mol. Sci. 2024, 25, 7949.
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, ID and other issues, individuals with FXS may exhibit sleep problems and impairment of circadian rhythm (CR). The Drosophila melanogaster models of FXS, such as dFMR1B55, present excellent model for research in the FXS field. During this study, sleep pattern and CR in dFMR1B55 mutants were analyzed, using a new platform based on continuous high-resolution videography in integration with a highly-customized version of 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 dFMR1 B55 male mutants 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 videography and software used in the current study should be recommended as a gold-standard methodology for such further research.
Keywords
Drosophila melanogaster model of fragile X syndrome; FMR1 gene; Fragile X syndrome; Circadian rhythm; Sleep.
Subject
Biology and Life Sciences, Behavioral Sciences
Copyright:
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