Article
Astrogliosis and neuroinflammation underlie scoliosis upon cilia dysfunction.
eLife - 10 Oct 2024
Djebar Morgane, Anselme Isabelle, Pezeron Guillaume, Bardet Pierre-Luc, Cantaut-Belarif Yasmine, Eschstruth Alexis, López-Santos Diego, Le Ribeuz Hélène, Jenett Arnim, Khoury Hanane, Veziers Joelle, Parmentier Caroline, Hirschler Aurélie, Carapito Christine, Bachmann-Gagescu Ruxandra, Schneider-Maunoury Sylvie, Vesque Christine
Abstract excerpt
Cilia defects lead to scoliosis in zebrafish, but the underlying pathogenic mechanisms are poorly understood and may diverge depending on the mutated gene. Here, we dissected the mechanisms of scoliosis onset in a zebrafish mutant for the rpgrip1l gene encoding a ciliary transition zone protein. rpgrip1l mutant fish developed scoliosis with near-total penetrance but asynchronous onset in juveniles. Taking...
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