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

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 appear to lack core components of the N...

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Publication status:
Published
Peer review status:
Peer reviewed
Version:
Publisher's version

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Publisher copy:
10.1242/jcs.194621

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Department:
Oxford, MSD, Pathology Dunn School
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Department:
Oxford, MSD, Biochemistry
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Department:
Oxford, MSD, Biochemistry
Barker, AR More by this author
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Funding agency for:
Titlow, JS
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Funding agency for:
Davis, I
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Funding agency for:
Raff, J
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Funding agency for:
Roque, H
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Publisher:
Company of Biologists Publisher's website
Journal:
Journal of Cell Science Journal website
Volume:
129
Issue:
20
Pages:
3732-3743
Publication date:
2016-09-30
Acceptance date:
2016-08-19
DOI:
ISSN:
0021-9533 and 1477-9137
Pubs id:
pubs:641764
URN:
uri:dc5900cd-ddb2-4d56-8610-dc60822fae47
UUID:
uuid:dc5900cd-ddb2-4d56-8610-dc60822fae47
Local pid:
pubs:641764
Language:
English
Keywords:

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