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Keywords: Cilia
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Journal Articles
Journal: Development
Development (2021) 148 (9): dev195552.
Published: 29 April 2021
...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...
Journal Articles
Journal: Development
Development (2021) 148 (4): dev192443.
Published: 17 February 2021
... 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2020) 147 (15): dev185579.
Published: 07 August 2020
.... , Stottmann , R. W. and Goto , J. ( 2018 ). A mutation in Ccdc39 causes neonatal hydrocephalus with abnormal motile cilia development in mice . Development (Camb.)   145 , dev154500 . 10.1242/dev.154500 Aboualaiwi , W. A. , Takahashi , M. , Mell , B. R. , Jones , T. J...
Journal Articles
Journal: Development
Development (2020) 147 (3): dev183301.
Published: 03 February 2020
.... 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2019) 146 (16): dev170985.
Published: 15 August 2019
... 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 on bilateral symmetry. They possess orthogonal anterior-posterior (A-P) and dorsal-ventral (D-V) axes...
Journal Articles
Journal: Development
Development (2019) 146 (6): dev172643.
Published: 15 March 2019
...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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2018) 145 (23): dev167130.
Published: 30 November 2018
... 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2018) 145 (23): dev159889.
Published: 28 November 2018
... 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2018) 145 (6): dev164236.
Published: 15 March 2018
... 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2018) 145 (1): dev154500.
Published: 09 January 2018
... 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2017) 144 (24): 4684–4693.
Published: 15 December 2017
..., 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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2017) 144 (8): 1544–1553.
Published: 15 April 2017
...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...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2017) 144 (2): 201–210.
Published: 15 January 2017
... contact the cerebral ventricles and bear a primary cilium. Although the role of the primary cilium in embryonic patterning has been studied, its role in brain ventricular morphogenesis is poorly characterized. Using conditional mutants, we show that the primary cilia of radial glia determine the size...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2016) 143 (24): 4654–4664.
Published: 15 December 2016
...Evan P. Campbell; Ian K. Quigley; Chris Kintner Multiciliated cell (MCC) differentiation involves extensive organelle biogenesis required to extend hundreds of motile cilia. Key transcriptional regulators known to drive the gene expression required for this organelle biogenesis are activated...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2015) 142 (23): 4068–4079.
Published: 01 December 2015
...S. Joseph Endicott; Basudha Basu; Mustafa Khokha; Martina Brueckner Vertebrate left-right (LR) asymmetry originates at a transient left-right organizer (LRO), a ciliated structure where cilia play a crucial role in breaking symmetry. However, much remains unknown about the choreography of cilia...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2015) 142 (19): 3429–3439.
Published: 01 October 2015
... biology. Among the processes that are organized by the core PCP proteins is the directional beating of cilia, such as those in the vertebrate node, airway and brain. Here, we exploit the live imaging capabilities of Xenopus to chart the progressive asymmetric localization of fluorescent reporters of Dvl1...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2015) 142 (13): 2291–2303.
Published: 01 July 2015
... and subsequently induction of more ventral cell fates. Furthermore, we demonstrate that activated Notch1 leads to pronounced accumulation of Smoothened (Smo) within primary cilia and elevated levels of full-length Gli3. Finally, we show that Notch activity promotes longer primary cilia both in vitro and in vivo...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2015) 142 (12): 2194–2202.
Published: 15 June 2015
... results in hair follicle morphogenesis defects due to attenuated Hh signaling; however, the loss of cilia makes it impossible to determine whether hair follicle phenotypes in these cilia mutants are caused by the loss of cilia, disruption of Hh signaling, or a combination of these events. In this study...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2015) 142 (3): 555–566.
Published: 01 February 2015
...Helen L. May-Simera; Ronald S. Petralia; Mireille Montcouquiol; Ya-Xian Wang; Katherine B. Szarama; Yun Liu; Weichun Lin; Michael R. Deans; Gregory J. Pazour; Matthew W. Kelley Primary cilia have been implicated in the generation of planar cell polarity (PCP). However, variations in the severity...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2014) 141 (23): 4558–4568.
Published: 01 December 2014
... along the ovary-uterus axis. In Celsr1 -deficient mice, the ciliary motion is not orchestrated along the ovary-uterus axis and the transport ability of beating cilia is impaired. Epithelial cells show less elongation and randomized orientation, and epithelial folds show randomized directionality...
Includes: Supplementary data