Publication: Astrogliosis and neuroinflammation underlie scoliosis upon cilia dysfunction
Astrogliosis and neuroinflammation underlie scoliosis upon cilia dysfunction
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Djebar, M., Anselme, I., Pezeron, G., Bardet, P.-L., Cantaut-Belarif, Y., Eschstruth, A., López-Santos, D., Le Ribeuz, H., Jenett, A., Khoury, H., Véziers, J., Parmentier, C., Hirschler, A., Carapito, C., Bachmann-Gagescu, R., Schneider-Maunoury, S., & Vesque, C. (2024). Astrogliosis and neuroinflammation underlie scoliosis upon cilia dysfunction. ELife, 13, RP96831. https://doi.org/10.7554/elife.96831
Abstract
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Abstract
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 advantage of this asynchrony, we found that curvature onset was preceded by ventricle dilations and was concomitant to the perturba
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- Djebar, Morgane
- Anselme, Isabelle
- Pezeron, Guillaume
- Bardet, Pierre-Luc
- Cantaut-Belarif, Yasmine
- Eschstruth, Alexis
- López-Santos, Diego
- Le Ribeuz, Hélène
- Jenett, Arnim
- Khoury, Hanane
- Véziers, Joelle
- Parmentier, Caroline
- Hirschler, Aurélie
- Carapito, Christine
- Schneider-Maunoury, Sylvie
- Vesque, Christine
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Djebar, M., Anselme, I., Pezeron, G., Bardet, P.-L., Cantaut-Belarif, Y., Eschstruth, A., López-Santos, D., Le Ribeuz, H., Jenett, A., Khoury, H., Véziers, J., Parmentier, C., Hirschler, A., Carapito, C., Bachmann-Gagescu, R., Schneider-Maunoury, S., & Vesque, C. (2024). Astrogliosis and neuroinflammation underlie scoliosis upon cilia dysfunction. ELife, 13, RP96831. https://doi.org/10.7554/elife.96831
