Ciliogenesis defects after neurulation impact brain development and neuronal activity in larval zebrafish

• Published in: iScience Vol. 27; no. 6; p. 110078
• Main Authors: D’Gama, Percival P., Jeong, Inyoung, Nygård, Andreas Moe, Trinh, Anh-Tuan, Yaksi, Emre, Jurisch-Yaksi, Nathalie
• Format: Journal Article
• Language: English
• Published: United States Elsevier Inc 21.06.2024; Elsevier
• Subjects: Biological sciences; Developmental neuroscience; Neuroscience; Neuroscience; Developmental neuroscience; Biological sciences
• ISSN: 2589-0042; 2589-0042
• DOI: 10.1016/j.isci.2024.110078

Cilia are slender, hair-like structures extending from cell surfaces and playing essential roles in diverse physiological processes. Within the nervous system, primary cilia contribute to signaling and sensory perception, while motile cilia facilitate cerebrospinal fluid flow. Here, we investigated the impact of ciliary loss on neural circuit development using a zebrafish line displaying ciliogenesis defects. We found that cilia defects after neurulation affect neurogenesis and brain morphology, especially in the cerebellum, and lead to altered gene expression profiles. Using whole brain calcium imaging, we measured reduced light-evoked and spontaneous neuronal activity in all brain regions. By shedding light on the intricate role of cilia in neural circuit formation and function in the zebrafish, our work highlights their evolutionary conserved role in the brain and sets the stage for future analysis of ciliopathy models. [Display omitted] •traf3ip1/elipsa mutant zebrafish display ciliogenesis defects after neurulation•Cilia defects lead to abnormal brain morphology, cell division, and gene expression•Cerebellar malformations and reduced number of Purkinje cells upon cilia defects•Reduced light-evoked and spontaneous neural activity in cilia mutants Biological sciences; Neuroscience; Developmental neuroscience