Abstract
The planar cell polarity (PCP) signaling pathway governs collective cell movements during vertebrate embryogenesis, and certain PCP proteins are also implicated in the assembly of cilia. The septins are cytoskeletal proteins controlling behaviors such as cell division and migration. Here, we identified control of septin localization by the PCP protein Fritz as a crucial control point for both collective cell movement and ciliogenesis in Xenopus embryos. We also linked mutations in human Fritz to Bardet-Biedl and Meckel-Gruber syndromes, a notable link given that other genes mutated in these syndromes also influence collective cell movement and ciliogenesis. These findings shed light on the mechanisms by which fundamental cellular machinery, such as the cytoskeleton, is regulated during embryonic development and human disease.
Publication types
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Research Support, N.I.H., Extramural
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Research Support, Non-U.S. Gov't
MeSH terms
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Abnormalities, Multiple / genetics
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Animals
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Bardet-Biedl Syndrome / genetics*
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Cell Membrane / metabolism
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Cell Membrane / ultrastructure
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Cell Movement*
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Cell Polarity*
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Cell Shape
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Cilia / metabolism*
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Cilia / ultrastructure
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Cytoskeletal Proteins / genetics
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Cytoskeletal Proteins / metabolism*
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Cytoskeleton / metabolism*
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Cytoskeleton / ultrastructure
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Embryo, Nonmammalian / cytology
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Embryo, Nonmammalian / physiology
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Embryonic Development
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Female
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GTP-Binding Proteins / genetics
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GTP-Binding Proteins / metabolism*
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Gastrula / cytology
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Genetic Association Studies
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Glycoproteins / genetics
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Glycoproteins / metabolism*
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Hedgehog Proteins / metabolism
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Humans
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Intracellular Signaling Peptides and Proteins
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Morphogenesis
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Mutant Proteins / metabolism
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Mutation
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Septins
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Syndrome
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Xenopus Proteins / genetics
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Xenopus Proteins / metabolism*
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Xenopus laevis
Substances
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Cytoskeletal Proteins
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Glycoproteins
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Hedgehog Proteins
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Intracellular Signaling Peptides and Proteins
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Mutant Proteins
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WD repeat containing planar cell polarity effector
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Xenopus Proteins
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GTP-Binding Proteins
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Septins
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sept2 protein, Xenopus
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sept7 protein, Xenopus