Universität zu Köln

Eguether, Thibaut ORCID: 0000-0003-1891-9253, Ermolaeva, Maria A., Zhao, Yongge, Bonnet, Marion C., Jain, Ashish, Pasparakis, Manolis ORCID: 0000-0002-9870-0966, Courtois, Gilles and Tassin, Anne-Marie (2014). The deubiquitinating enzyme CYLD controls apical docking of basal bodies in ciliated epithelial cells. Nat. Commun., 5. LONDON: NATURE PUBLISHING GROUP. ISSN 2041-1723

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Abstract

CYLD is a tumour suppressor gene mutated in familial cylindromatosis, a genetic disorder leading to the development of skin appendage tumours. It encodes a deubiquitinating enzyme that removes Lys63-or linear-linked ubiquitin chains. CYLD was shown to regulate cell proliferation, cell survival and inflammatory responses, through various signalling pathways. Here we show that CYLD localizes at centrosomes and basal bodies via interaction with the centrosomal protein CAP350 and demonstrate that CYLD must be both at the centrosome and catalytically active to promote ciliogenesis independently of NF-kB. In transgenic mice engineered to mimic the smallest truncation found in cylindromatosis patients, CYLD interaction with CAP350 is lost disrupting CYLD centrosome localization, which results in cilia formation defects due to impairment of basal body migration and docking. These results point to an undiscovered regulation of ciliogenesis by Lys63 ubiquitination and provide new perspectives regarding CYLD function that should be considered in the context of cylindromatosis.

Item Type:	Journal Article
Creators:	Creators Email ORCID ORCID Put Code Eguether, Thibaut UNSPECIFIED orcid.org/0000-0003-1891-9253 UNSPECIFIED Ermolaeva, Maria A. UNSPECIFIED UNSPECIFIED UNSPECIFIED Zhao, Yongge UNSPECIFIED UNSPECIFIED UNSPECIFIED Bonnet, Marion C. UNSPECIFIED UNSPECIFIED UNSPECIFIED Jain, Ashish UNSPECIFIED UNSPECIFIED UNSPECIFIED Pasparakis, Manolis UNSPECIFIED orcid.org/0000-0002-9870-0966 UNSPECIFIED Courtois, Gilles UNSPECIFIED UNSPECIFIED UNSPECIFIED Tassin, Anne-Marie UNSPECIFIED UNSPECIFIED UNSPECIFIED
URN:	urn:nbn:de:hbz:38-432550
DOI:	10.1038/ncomms5585
Journal or Publication Title:	Nat. Commun.
Volume:	5
Date:	2014
Publisher:	NATURE PUBLISHING GROUP
Place of Publication:	LONDON
ISSN:	2041-1723
Language:	English
Faculty:	Faculty of Mathematics and Natural Sciences
Divisions:	Faculty of Mathematics and Natural Sciences > Department of Biology > Institute for Genetics
Subjects:	no entry
Uncontrolled Keywords:	Keywords Language NF-KAPPA-B; PRIMARY CILIUM; CENTROSOMAL PROTEINS; GOLGI-COMPLEX; CILIOGENESIS; CENTRIOLES; DOMAIN; EB1; CAP350; DIFFERENTIATION Multiple languages Multidisciplinary Sciences Multiple languages
Refereed:	Yes
URI:	http://kups.ub.uni-koeln.de/id/eprint/43255