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Gegg, M. ; Böttcher, A. ; Burtscher, I. ; Hasenöder, S. ; van Campenhout, C.A.* ; Aichler, M. ; Walch, A.K. ; Grant, S.G.* ; Lickert, H.

Flattop regulates basal body docking and positioning in mono- and multiciliated cells.

eLife 3:e03842 (2014)
Publ. Version/Full Text DOI PMC
Open Access Gold
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Planar cell polarity (PCP) regulates basal body (BB) docking and positioning during cilia formation, but the underlying mechanisms remain elusive. Here, we investigate the uncharacterized gene Flattop (Fltp) that is transcriptionally activated during PCP acquisition in ciliated tissues. Fltp knock-out mice show BB docking and ciliogenesis defects in multiciliated lung cells. Furthermore, Fltp is necessary for kinocilium positioning in monociliated inner ear hair cells. In these cells, the core PCP molecule Dishevelled 2, the BB/spindle positioning protein Dlg3 and Fltp localize directly adjacent at the apical plasma membrane, physically interact and surround the BB at the interface of the microtubule and actin cytoskeleton. Dlg3 and Fltp knock-outs suggest that both cooperatively translate PCP cues for BB positioning in the inner ear. Taken together, the identification of novel BB/spindle positioning components as potential mediators of PCP signaling might have broader implications for other cell types, ciliary disease and asymmetric cell division.
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Publication type Article: Journal article
Document type Scientific Article
Keywords Flattop ; Actin ; Basal Body ; Cell Biology ; Developmental Biology ; Microtubule ; Mouse ; Planar Cell Polarity ; Spindle Positioning ; Stem Cells
Language english
Publication Year 2014
HGF-reported in Year 2014
ISSN (print) / ISBN 2050-084X
e-ISSN 2050-084X
Journal eLife
Quellenangaben Volume: 3, Issue: , Pages: , Article Number: e03842 Supplement: ,
Publisher eLife Sciences Publications
Reviewing status Peer reviewed
Institute(s) Institute of Diabetes and Regeneration Research (IDR)
Research Unit Analytical Pathology (AAP)
Institute of Pathology (PATH)
POF-Topic(s) 30201 - Metabolic Health
90000 - German Center for Diabetes Research
30205 - Bioengineering and Digital Health
30504 - Mechanisms of Genetic and Environmental Influences on Health and Disease
Research field(s) Helmholtz Diabetes Center
Enabling and Novel Technologies
PSP Element(s) G-502300-001
G-501900-231
G-500390-001
G-500300-001
PubMed ID 25296022
DOI 10.7554/eLife.03842
WOS ID WOS:000343418100001
Erfassungsdatum 2014-10-10
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