...Mycah T. Sewell; Emilie Legué; Karel F. Liem, Jr ABSTRACT Cilia are microtubule (MT)-based organelles present on the surface of nearly all vertebrate cells. MTs are polymers of α- and β-tubulins that are each encoded by multiple, individual isotype genes. Tubulin isotype composition is thought...

... cells ablated of centrosomes. Finally, quantification of Cep120, centrosome and cilia number showed a significant reduction in cystic epithelial cells of Cep120 KO mice at P15 ( Fig. S6B-E ). Several of the pathways/processes identified in the Hallmark and CompBio analyses are downstream effectors...

... of tubulin-related biology in autism risk. Autism spectrum disorder Microtubules Tubulin Spindles Cilia Chromatin Cell cycle Xenopus Human Overlook International Foundation National Institute of Mental Health http://dx.doi.org/10.13039/100000025 1U01MH115747-01A1...

...Hannah M. Wesselman; Ana L. Flores-Mireles; Aidan Bauer; Liming Pei; Rebecca A. Wingert ABSTRACT Cilia are essential for the ontogeny and function of many tissues, including the kidney. Here, we report that transcription factor ERRγ ortholog estrogen related receptor gamma a (Esrrγa) is essential...

... Neuron Evolution Cilia Cnidaria Nematostella vectensis Natural Environment Research Council http://dx.doi.org/10.13039/501100000270 Marine Biological Association http://dx.doi.org/10.13039/501100000655 Austrian Science Fund http://dx.doi.org/10.13039/501100002428...

... associated microtubule organization may play a role in correct positioning of the basal body for left-right symmetry breaking. Basal body Cilia Left-right asymmetry Planar cell polarity Mouse Ministry of Education, Culture, Sports, Science, and Technology 17H01435 Core...

... to be dependent on cilia-driven fluid flow in the embryonic node. Planar cell polarity (PCP) signalling is crucial for patterning of nodal cilia, yet downstream effectors driving this process remain elusive. We have examined the role of the JNK gene family, a proposed downstream component of PCP signalling...

...Siyu Liu; Jintao Zhang; Zine Eddine Kherraf; Shuya Sun; Xin Zhang; Caroline Cazin; Charles Coutton; Raoudha Zouari; Shuqin Zhao; Fan Hu; Selima Fourati Ben Mustapha; Christophe Arnoult; Pierre F. Ray; Mingxi Liu ABSTRACT Defects in the structure or motility of cilia and flagella may lead to severe...

...Emily K. Ho; Tim Stearns ABSTRACT The mechanisms of vertebrate Hedgehog signaling are linked to the biology of the primary cilium, an antenna-like organelle that projects from the surface of most vertebrate cell types. Although the advantages of restricting signal transduction to cilia are often...

... in zebrafish. Additionally, PKA-mediated phosphorylation of Gpr161 controls its ciliary localisation to fine-tune Hedgehog signalling activity. Hedgehog signalling Gpr161 PKA Cilia Zebrafish The Hh signalling pathway is a key regulator of cell fate specification and proliferation during...

... and transducing flow during embryogenesis. Shear stress Blood Lymph Cerebrospinal fluid Cilia Mechanosensation Fonds Wetenschappelijk Onderzoek 10.13039/501100003130 11ZD516N G091018N G0B5920N KU Leuven 10.13039/501100004040 C14/19/095 Horizon 2020 10.13039...

.... Cilia are essential for Shh signal transduction and the ciliary phosphatidylinositol phosphatase Inpp5e is linked to Shh regulation. In the course of a forward genetic screen for recessive mouse mutants, we identified a functional null allele of inositol polyphosphate-5-phosphatase E ( Inpp5e ), ridge...

... of asymmetric and symmetric structures. This Review discusses how symmetries and asymmetries can be generated alongside each other in the embryo and then maintained during growth. Symmetry Asymmetry Nodal Left-right Limbs Somites Cilia The vast majority of animals have body plans based...

...Hao Lu; Priyanka Anujan; Feng Zhou; Yiliu Zhang; Yan Ling Chong; Colin D. Bingle; Sudipto Roy ABSTRACT Motile cilia on multiciliated cells (MCCs) function in fluid clearance over epithelia. Studies with Xenopus embryos and individuals with the congenital respiratory disorder reduced generation...

... of the primary mesenchyme cells, gut morphology, and cilia. These defects likely result from increased Dvl protein levels, leading to perturbation of Wnt-dependent signaling pathways and additional Dvl-mediated processes. We further demonstrated that overexpression of Dvl isoforms recapitulated some of the Dvl...

... patterning. First, WDR5 is expressed in the nuclei of monociliated cells of the LR organizer (LRO) and regulates foxj1 expression. LR defects in wdr5 morphants can be partially rescued with the addition of foxj1 . Second, WDR5 localizes to the bases of cilia. Using a mutant form of WDR5, we demonstrate...

... the transition zone (TZ). The TZ serves as a gatekeeper to control protein composition in cilia, but less is known about its role in ciliary bud formation. Here, we show that centrosomal protein Dzip1l is required for Hh signaling between Smoothened and Sufu. Dzip1l colocalizes with basal body appendage proteins...

... postnatal hydrocephalus in the progressive hydrocephal us ( prh ) mouse mutant. Ccdc39 is selectively expressed in embryonic choroid plexus and ependymal cells on the medial wall of the forebrain ventricle, and the protein is localized to the axoneme of motile cilia. The Ccdc39 prh/prh ependymal cells...

..., we show that Pontin is essential for cilia motility in both zebrafish and mouse and that Pontin and Reptin function cooperatively in this process. Zebrafish pontin mutants display phenotypes tightly associated with cilia defects, and cilia motility is lost in a number of ciliated tissues along...

...Daisy Xin; Kasey J. Christopher; Lewie Zeng; Yong Kong; Scott D. Weatherbee Cilia are key regulators of animal development and depend on intraflagellar transport (IFT) proteins for their formation and function, yet the roles of individual IFT proteins remain unclear. We examined the Ift56 hop mouse...