# Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System

**Authors:** Sara Milojevic, Arijit Ghosh, Vedrana Makevic, Maja Stojkovic, Maria Capovilla, Tomislav Tosti, Dejan Budimirovic, Dragana Protic

PMC · DOI: 10.3390/ijms25147949 · International Journal of Molecular Sciences · 2024-07-20

## TL;DR

This study uses fruit flies to investigate sleep and circadian rhythm issues in a model of Fragile X Syndrome, using a new video-based system for more accurate results.

## Contribution

The study introduces a new video-based system for analyzing sleep and circadian rhythms in fruit fly models of Fragile X Syndrome.

## Key findings

- dFMR1B55 male mutants sleep more and show weak rhythmicity rather than being completely arrhythmic.
- The new video-based system provides more sensitive and accurate results for behavioral monitoring in fruit flies.
- The method is affordable and can be widely adopted for high-resolution behavior studies in genetic disorder models.

## 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.

## Linked entities

- **Genes:** FMR1 (fragile X messenger ribonucleoprotein 1) [NCBI Gene 2332]
- **Diseases:** Fragile X Syndrome (MONDO:0010383), autism spectrum disorder (MONDO:0005258)
- **Species:** Drosophila melanogaster (taxon 7227)

## Full-text entities

- **Genes:** Fmr1 (Fragile X messenger ribonucleoprotein 1) [NCBI Gene 37528] {aka AT24755, BcDNA:GM08679, CG6203, DFMRP, DFmr1, Dfmr}
- **Diseases:** impairment of circadian rhythm (MESH:D021081), ID (MESH:D008607), FXS (MESH:D005600), ASD (MESH:D000067877), sleep problems (MESH:D012893), genetic disorder (MESH:D030342), arrhythmic (OMIM:212500)
- **Species:** Drosophila melanogaster (fruit fly, species) [taxon 7227]

## Full text

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## Figures

5 figures with captions in the complete paper: https://tomesphere.com/paper/PMC11277495/full.md

## References

58 references — full list in the complete paper: https://tomesphere.com/paper/PMC11277495/full.md

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Source: https://tomesphere.com/paper/PMC11277495