{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T01:17:51Z","timestamp":1760145471032,"version":"build-2065373602"},"reference-count":58,"publisher":"MDPI AG","issue":"14","license":[{"start":{"date-parts":[[2024,7,20]],"date-time":"2024-07-20T00:00:00Z","timestamp":1721433600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Science Fund of the Republic of Serbia","award":["7673781","451-03-66\/2024-03\/200110","451-03-66\/2024-03\/200026"],"award-info":[{"award-number":["7673781","451-03-66\/2024-03\/200110","451-03-66\/2024-03\/200026"]}]},{"name":"Ministry of Science, Technological Development and Innovation of the Republic of Serbia","award":["7673781","451-03-66\/2024-03\/200110","451-03-66\/2024-03\/200026"],"award-info":[{"award-number":["7673781","451-03-66\/2024-03\/200110","451-03-66\/2024-03\/200026"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>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.<\/jats:p>","DOI":"10.3390\/ijms25147949","type":"journal-article","created":{"date-parts":[[2024,7,23]],"date-time":"2024-07-23T07:57:23Z","timestamp":1721721443000},"page":"7949","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Circadian Rhythm and Sleep Analyses in a Fruit Fly Model of Fragile X Syndrome Using a Video-Based Automated Behavioral Research System"],"prefix":"10.3390","volume":"25","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8280-3015","authenticated-orcid":false,"given":"Sara","family":"Milojevic","sequence":"first","affiliation":[{"name":"Department of Pharmacology, Clinical Pharmacology and Toxicology, Faculty of Medicine, University of Belgrade, 11000 Belgrade, Serbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7910-3170","authenticated-orcid":false,"given":"Arijit","family":"Ghosh","sequence":"additional","affiliation":[{"name":"Chronobiology and Behavioral Neurogenetics Laboratory, Neuroscience Unit, Jawaharlal Nehru Center for Advanced Scientific Research, Jakkur, Bangalore 560064, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vedrana","family":"Makevic","sequence":"additional","affiliation":[{"name":"Department of Pathophysiology, Faculty of Medicine, University of Belgrade, 11000 Belgrade, Serbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0706-3800","authenticated-orcid":false,"given":"Maja","family":"Stojkovic","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, Clinical Pharmacology and Toxicology, Faculty of Medicine, University of Belgrade, 11000 Belgrade, Serbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1308-7964","authenticated-orcid":false,"given":"Maria","family":"Capovilla","sequence":"additional","affiliation":[{"name":"UMR7275 CNRS-INSERM-UniCA, Institute of Cellular and Molecular Pharmacology Institute, Sophia Antipolis, 06560 Valbonne, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1403-4688","authenticated-orcid":false,"given":"Tomislav","family":"Tosti","sequence":"additional","affiliation":[{"name":"Institute of Chemistry, Technology and Metallurgy, National Institute of the Republic of Serbia, University of Belgrade, 11000 Belgrade, Serbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7263-5134","authenticated-orcid":false,"given":"Dejan","family":"Budimirovic","sequence":"additional","affiliation":[{"name":"Department of Psychiatry, Fragile X Clinic, Kennedy Krieger Institute, Baltimore, MD 21205, USA"},{"name":"Department of Psychiatry & Behavioral Sciences-Child Psychiatry, School of Medicine, Johns Hopkins University, Baltimore, MD 21205, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2137-5405","authenticated-orcid":false,"given":"Dragana","family":"Protic","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, Clinical Pharmacology and Toxicology, Faculty of Medicine, University of Belgrade, 11000 Belgrade, Serbia"},{"name":"Fragile X Clinic, Special Hospital for Cerebral Palsy and Developmental Neurology, 11000 Belgrade, Serbia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2024,7,20]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"905","DOI":"10.1016\/0092-8674(91)90397-H","article-title":"Identification of a Gene (FMR-1) Containing a CGG Repeat Coincident with a Breakpoint Cluster Region Exhibiting Length Variation in Fragile X Syndrome","volume":"65","author":"Verkerk","year":"1991","journal-title":"Cell"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1001","DOI":"10.1016\/0092-8674(95)90279-1","article-title":"Cloning and Functional Analysis of TipE, a Novel Membrane Protein That Enhances Drosophila Para Sodium Channel Function","volume":"82","author":"Feng","year":"1995","journal-title":"Cell"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"5395","DOI":"10.1073\/pnas.94.10.5395","article-title":"Fragile X Mental Retardation Protein Is Translated near Synapses in Response to Neurotransmitter Activation","volume":"94","author":"Weiler","year":"1997","journal-title":"Proc. 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