Universität zu Köln

Falk, Nathalie ORCID: 0000-0001-6722-0292, Kessler, Kristin, Schramm, Sinja-Fee, Boldt, Karsten, Becirovic, Elvir ORCID: 0000-0001-8801-0649, Michalakis, Stylianos ORCID: 0000-0001-5092-9238, Regus-Leidig, Hanna, Noegel, Angelika A., Ueffing, Marius, Thiel, Christian T., Roepman, Ronald ORCID: 0000-0002-5178-8163, Brandstaetter, Johann Helmut and Giessl, Andreas ORCID: 0000-0002-9768-2049 (2018). Functional analyses of Pericentrin and Syne-2 interaction in ciliogenesis. J. Cell Sci., 131 (16). CAMBRIDGE: COMPANY BIOLOGISTS LTD. ISSN 1477-9137

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Abstract

Pericentrin (Pcnt) is a multifunctional scaffold protein and mutations in the human PCNT gene are associated with several diseases, including ciliopathies. Pcnt plays a crucial role in ciliary development in olfactory receptor neurons, but its function in the photoreceptorconnecting cilium is unknown. We downregulated Pcnt in the retina ex vivo and in vivo via a virus-based RNA interference approach to study Pcnt function in photoreceptors. ShRNA-mediated knockdown of Pcnt impaired the development of the connecting cilium and the outer segment of photoreceptors, and caused a nuclear migration defect. In protein interaction screens, we found that the outer nuclear membrane protein Syne-2 (also known as Nesprin-2) is an interaction partner of Pcnt in photoreceptors. Syne-2 is important for positioning murine photoreceptor cell nuclei and for centrosomal migration during early ciliogenesis. CRISPR/Cas9mediated knockout of Syne-2 in cell culture led to an overexpression and mislocalization of Pcnt and to ciliogenesis defects. Our findings suggest that the Pcnt-Syne-2 complex is important for ciliogenesis and outer segment formation during retinal development and plays a role in nuclear migration.

Item Type:	Journal Article
Creators:	Creators Email ORCID ORCID Put Code Falk, Nathalie UNSPECIFIED orcid.org/0000-0001-6722-0292 UNSPECIFIED Kessler, Kristin UNSPECIFIED UNSPECIFIED UNSPECIFIED Schramm, Sinja-Fee UNSPECIFIED UNSPECIFIED UNSPECIFIED Boldt, Karsten UNSPECIFIED UNSPECIFIED UNSPECIFIED Becirovic, Elvir UNSPECIFIED orcid.org/0000-0001-8801-0649 UNSPECIFIED Michalakis, Stylianos UNSPECIFIED orcid.org/0000-0001-5092-9238 UNSPECIFIED Regus-Leidig, Hanna UNSPECIFIED UNSPECIFIED UNSPECIFIED Noegel, Angelika A. UNSPECIFIED UNSPECIFIED UNSPECIFIED Ueffing, Marius UNSPECIFIED UNSPECIFIED UNSPECIFIED Thiel, Christian T. UNSPECIFIED UNSPECIFIED UNSPECIFIED Roepman, Ronald UNSPECIFIED orcid.org/0000-0002-5178-8163 UNSPECIFIED Brandstaetter, Johann Helmut UNSPECIFIED UNSPECIFIED UNSPECIFIED Giessl, Andreas UNSPECIFIED orcid.org/0000-0002-9768-2049 UNSPECIFIED
URN:	urn:nbn:de:hbz:38-177262
DOI:	10.1242/jcs.218487
Journal or Publication Title:	J. Cell Sci.
Volume:	131
Number:	16
Date:	2018
Publisher:	COMPANY BIOLOGISTS LTD
Place of Publication:	CAMBRIDGE
ISSN:	1477-9137
Language:	English
Faculty:	Unspecified
Divisions:	Unspecified
Subjects:	no entry
Uncontrolled Keywords:	Keywords Language CENTROSOME PROTEIN; NUCLEAR MIGRATION; COMPLEXES; ARCHITECTURE; MUTATIONS; NESPRIN-2; ENVELOPE; CILIUM; GOLGI; LOCALIZATION Multiple languages Cell Biology Multiple languages
Refereed:	Yes
URI:	http://kups.ub.uni-koeln.de/id/eprint/17726