Ciliogenesis defects after neurulation impact brain development and neuronal activity in larval zebrafish
Percival P. D’Gama,
Inyoung Jeong,
Andreas Moe Nygård,
Anh-Tuan Trinh,
Emre Yaksi,
Nathalie Jurisch-Yaksi
Affiliations
Percival P. D’Gama
Department of Clinical and Molecular Medicine, Norwegian University of Science and Technology, Erling Skalgssons gate 1, 7030 Trondheim, Norway
Inyoung Jeong
Department of Clinical and Molecular Medicine, Norwegian University of Science and Technology, Erling Skalgssons gate 1, 7030 Trondheim, Norway
Andreas Moe Nygård
Department of Clinical and Molecular Medicine, Norwegian University of Science and Technology, Erling Skalgssons gate 1, 7030 Trondheim, Norway
Anh-Tuan Trinh
Kavli Institute for Systems Neuroscience and Centre for Algorithms in the Cortex, Norwegian University of Science and Technology, Olav Kyrres Gate 9, 7030 Trondheim, Norway
Emre Yaksi
Kavli Institute for Systems Neuroscience and Centre for Algorithms in the Cortex, Norwegian University of Science and Technology, Olav Kyrres Gate 9, 7030 Trondheim, Norway; Koç University Research Center for Translational Medicine, Koç University School of Medicine, Davutpaşa Caddesi, No:4, Topkapı 34010, Istanbul, Turkey
Nathalie Jurisch-Yaksi
Department of Clinical and Molecular Medicine, Norwegian University of Science and Technology, Erling Skalgssons gate 1, 7030 Trondheim, Norway; Kavli Institute for Systems Neuroscience and Centre for Algorithms in the Cortex, Norwegian University of Science and Technology, Olav Kyrres Gate 9, 7030 Trondheim, Norway; Corresponding author
Summary: 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.
