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Drosophila sensory cilia lacking MKS-proteins exhibit striking defects during development but only subtle defects in adults

Metta B. Pratt, Joshua S. Titlow, Ilan Davis, Amy R. Barker, Helen R. Dawe, Jordan W. Raff, Helio Roque
doi: https://doi.org/10.1101/058081
Metta B. Pratt
1The Sir William Dunn School of Pathology, University of Oxford, South Parks Road, Oxford OX1 3RE
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Joshua S. Titlow
2Department of Biochemistry, University of Oxford, South Parks Road, Oxford OX1 3QU
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Ilan Davis
2Department of Biochemistry, University of Oxford, South Parks Road, Oxford OX1 3QU
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Amy R. Barker
3Biosciences, College of Life and Environmental Sciences, University of Exeter, Exeter EX4 4QD
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Helen R. Dawe
3Biosciences, College of Life and Environmental Sciences, University of Exeter, Exeter EX4 4QD
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Jordan W. Raff
1The Sir William Dunn School of Pathology, University of Oxford, South Parks Road, Oxford OX1 3RE
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  • For correspondence: jordan.raff@path.ox.ac.uk helio.roque@path.ox.ac.uk
Helio Roque
1The Sir William Dunn School of Pathology, University of Oxford, South Parks Road, Oxford OX1 3RE
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  • For correspondence: jordan.raff@path.ox.ac.uk helio.roque@path.ox.ac.uk
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Abstract

Cilia are conserved organelles that have important motility, sensory and signalling roles. The transition zone (TZ) at the base of the cilium is critical for cilia function, and defects in several TZ proteins are associated with human congenital ciliopathies such as Nephronophthisis (NPHP) and Meckel Gruber syndrome (MKS). In several species, MKS and NPHP proteins form separate complexes that cooperate with Cep290 to assemble the TZ, but flies lack an obvious NPHP complex. We show that MKS proteins in flies are spatially separated from Cep290 at the TZ, and that flies mutant for individual MKS genes fail to recruit other MKS proteins to the TZ, while Cep290 appears to be recruited normally. Although there are abnormalities in microtubule and membrane organisation in developing MKS mutant cilia, these defects are less apparent in adults, where sensory cilia and sperm flagella function quite normally. Thus, surprisingly, MKS proteins are not essential for cilia or flagella function in flies.

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The copyright holder for this preprint is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under a CC-BY-ND 4.0 International license.
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Posted June 10, 2016.
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Drosophila sensory cilia lacking MKS-proteins exhibit striking defects during development but only subtle defects in adults
Metta B. Pratt, Joshua S. Titlow, Ilan Davis, Amy R. Barker, Helen R. Dawe, Jordan W. Raff, Helio Roque
bioRxiv 058081; doi: https://doi.org/10.1101/058081
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Drosophila sensory cilia lacking MKS-proteins exhibit striking defects during development but only subtle defects in adults
Metta B. Pratt, Joshua S. Titlow, Ilan Davis, Amy R. Barker, Helen R. Dawe, Jordan W. Raff, Helio Roque
bioRxiv 058081; doi: https://doi.org/10.1101/058081

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