| Literature DB >> 30096282 |
Helen Rankin Willsey1, Peter Walentek2, Cameron R T Exner3, Yuxiao Xu1, Andrew B Lane4, Richard M Harland4, Rebecca Heald4, Niovi Santama5.
Abstract
Microtubule remodeling is critical for cellular and developmental processes underlying morphogenetic changes and for the formation of many subcellular structures. Katanins are conserved microtubule severing enzymes that are essential for spindle assembly, ciliogenesis, cell division, and cellular motility. We have recently shown that a related protein, Katanin-like 2 (KATNAL2), is similarly required for cytokinesis, cell cycle progression, and ciliogenesis in cultured mouse cells. However, its developmental expression pattern, localization, and in vivo role during organogenesis have yet to be characterized. Here, we used Xenopus embryos to reveal that Katnal2 (1) is expressed broadly in ciliated and neurogenic tissues throughout embryonic development; (2) is localized to basal bodies, ciliary axonemes, centrioles, and mitotic spindles; and (3) is required for ciliogenesis and brain development. Since human KATNAL2 is a risk gene for autism spectrum disorders, our functional data suggest that Xenopus may be a relevant system for understanding the relationship of mutations in this gene to autism and the underlying molecular mechanisms of pathogenesis.Entities:
Keywords: Autism; Cilia; Katanin; Katnal2; Neurogenesis; Xenopus
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Year: 2018 PMID: 30096282 PMCID: PMC6143417 DOI: 10.1016/j.ydbio.2018.08.002
Source DB: PubMed Journal: Dev Biol ISSN: 0012-1606 Impact factor: 3.582