biostudies-literatureUnknown11(10)Dona MCiliopathies are Mendelian disorders caused by dysfunction of cilia, ubiquitous organelles involved in fluid propulsion (motile cilia) or signal transduction (primary cilia). Retinal dystrophy is a common phenotypic characteristic of ciliopathies since photoreceptor outer segments are specialized primary cilia. These ciliary structures heavily rely on intracellular minus-end directed transport of cargo, mediated at least in part by the cytoplasmic dynein 1 motor complex, for their formation, maintenance and function. Ninein-like protein (NINL) is known to associate with this motor complex and is an important interaction partner of the ciliopathy-associated proteins lebercilin, USH2A and CC2D2A. Here, we scrutinize the function of NINL with combined proteomic and zebrafish in vivo approachePLoS geneticse1005574https://www.ebi.ac.uk/biostudies/studies/S-EPMC4617706biostudies-literatureNINL and DZANK1 Co-function in Vesicle Transport and Are Essential for Photoreceptor Development in Zebrafish.PMC4617706Gibson TJvan Beersum SEKremer HPeters TATonnaer ELvan Wijk EUeffing MRoepman RKeunen JEBachmann-Gagescu RSlijkerman RWDona MToedt Gvan Reeuwijk JHetterschijt LBergboer JGFlik Gde Vrieze ETexier YBoldt KHorn NfalseNINL and DZANK1 Co-function in Vesicle Transport and Are Essential for Photoreceptor Development in Zebrafish.Ciliopathies are Mendelian disorders caused by dysfunction of cilia, ubiquitous organelles involved in fluid propulsion (motile cilia) or signal transduction (primary cilia). Retinal dystrophy is a common phenotypic characteristic of ciliopathies since photoreceptor outer segments are specialized primary cilia. These ciliary structures heavily rely on intracellular minus-end directed transport of cargo, mediated at least in part by the cytoplasmic dynein 1 motor complex, for their formation, maintenance and function. Ninein-like protein (NINL) is known to associate with this motor complex and is an important interaction partner of the ciliopathy-associated proteins lebercilin, USH2A and CC2D2A. Here, we scrutinize the function of NINL with combined proteomic and zebrafish in vivo approache2015-01-01T00:00:00Z2015 Oct2026-04-16T21:15:39.617Z2019-03-26T22:46:02ZS-EPMC46177062648551410.1371/journal.pgen.1005574