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Mitochondrial dysfunctions are associated with neurodevelopmental disorders including Fragile X syndrome (FXS), the most common cause of inherited intellectual disability, which also presents with motor skill deficits. However, the precise role of mitochondria in the pathophysiology of FXS remains largely unknown. Notably, previous studies have linked the serotonergic system and mitochondrial activity to FXS. Our study investigates the potential therapeutic role of serotonin receptor 1A (5-HT1A) in FXS. Using the Drosophila model of FXS, we demonstrated that treatment with eltoprazine, a 5-HT1A agonist, can ameliorate synaptic transmission, correct mitochondrial deficits, and ultimately improve motor behavior. While these findings suggest that the 5-HT1A-mitochondrial axis may be a promising therapeutic target, further investigation is needed in the context of FXS.<\/jats:p>","DOI":"10.3390\/ijms25168787","type":"journal-article","created":{"date-parts":[[2024,8,14]],"date-time":"2024-08-14T05:23:27Z","timestamp":1723613007000},"page":"8787","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Activation of the 5-HT1A Receptor by Eltoprazine Restores Mitochondrial and Motor Deficits in a Drosophila Model of Fragile X Syndrome"],"prefix":"10.3390","volume":"25","author":[{"given":"Anna","family":"Vannelli","sequence":"first","affiliation":[{"name":"Department of Fundamental Neurosciences, University of Lausanne, 1005 Lausanne, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9848-0262","authenticated-orcid":false,"given":"Vittoria","family":"Mariano","sequence":"additional","affiliation":[{"name":"Department of Fundamental Neurosciences, University of Lausanne, 1005 Lausanne, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Claudia","family":"Bagni","sequence":"additional","affiliation":[{"name":"Department of Fundamental Neurosciences, University of Lausanne, 1005 Lausanne, Switzerland"},{"name":"Department of Biomedicine and Prevention, University of Rome Tor Vergata, 00133 Rome, Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2094-7491","authenticated-orcid":false,"given":"Alexandros K.","family":"Kanellopoulos","sequence":"additional","affiliation":[{"name":"Department of Fundamental Neurosciences, University of Lausanne, 1005 Lausanne, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2024,8,13]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"C641","DOI":"10.1152\/ajpcell.00222.2006","article-title":"Mitochondria and Neuronal Activity","volume":"292","author":"Kann","year":"2006","journal-title":"AJP Cell Physiol."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"671","DOI":"10.1242\/jcs.171017","article-title":"Mitochondrial Dynamics in Neuronal Injury, Development and Plasticity","volume":"130","author":"Flippo","year":"2017","journal-title":"J. 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