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Keywords: Primary cilia
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Neural development
Journal: Development
Development (2022) 149 (21): dev199931.
Published: 31 October 2022
... manner, as well as defects in primary cilia. Furthermore, loss of PAR3 enhanced ligand-independent ciliary accumulation of smoothened and an inhibitor of smoothened ameliorated the hyperproliferation of NPCs in the telencephalon. Thus, these findings support the idea that PAR3 has a crucial role...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2021) 148 (4): dev194175.
Published: 15 February 2021
...Christian Louis Bonatto Paese; Evan C. Brooks; Megan Aarnio-Peterson; Samantha A. Brugmann ABSTRACT Ciliopathies represent a growing class of diseases caused by defects in microtubule-based organelles called primary cilia. Approximately 30% of ciliopathies are characterized by craniofacial...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2018) 145 (18): dev151407.
Published: 17 September 2018
..., and the cilium can reside at the cell surface or assume a more submerged position (for a review, see Bernabé-Rubio and Alonso, 2017 ). For example, in mammals, the primary cilia in epithelial cells of kidney tubules are at the cell surface ( Latta et al., 1961 ), whereas those in smooth muscle cells...
Journal Articles
Journal: Development
Development (2018) 145 (1): dev154054.
Published: 8 January 2018
... understood. The orphan G protein-coupled receptor Gpr161 localizes to primary cilia and functions as a negative regulator of Shh signaling by promoting Gli transcriptional repressor versus activator formation. Here, we show that forelimb buds are not formed in Gpr161 knockout mouse embryos despite...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2016) 143 (12): 2160–2171.
Published: 15 June 2016
...Laura Grisanti; Ekaterina Revenkova; Ronald E. Gordon; Carlo Iomini Primary cilia have been linked to signaling pathways involved in cell proliferation, cell motility and cell polarity. Defects in ciliary function result in developmental abnormalities and multiple ciliopathies. Patients affected...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2016) 143 (9): 1491–1501.
Published: 1 May 2016
... birth owing to organogenesis defects as in ciliopathies. Shh signaling is impaired in null embryos and primary cilia are reduced in multiple tissues. We show that CP110 is required for anchoring of basal bodies to the membrane during cilia formation. CP110 loss resulted in an abnormal distribution...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2015) 142 (23): 4080–4091.
Published: 1 December 2015
... ) in the endocardium to promote trabeculation and that forced Notch activation in the absence of cardiac contraction rescues efnb2a and nrg1 expression. Using in vitro and in vivo systems, we showed that primary cilia are important mediators of fluid flow to stimulate Notch expression. Together, our findings describe...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2014) 141 (15): 3003–3012.
Published: 1 August 2014
.... Although phenotypically similar to talpid 3 , talpid 2 has a distinct facial phenotype and an unknown cellular, molecular and genetic basis. We set out to determine the etiology of the craniofacial phenotype of this mutant. We confirmed that primary cilia were disrupted in talpid 2 mutants. Molecularly, we...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2011) 138 (15): 3261–3272.
Published: 1 August 2011
... regions, depending on hedgehog (Hh) signalling, and exhibits embryonic lethality. The talpid 3 chicken has a mutation in KIAA0586 , which encodes a centrosomal protein required for the formation of primary cilia, which are sites of vertebrate Hh signalling. The highly conserved exons 11 and 12 of KIAA0586...
Includes: Multimedia, Supplementary data
Journal Articles
Journal: Development
Development (2009) 136 (4): 655–664.
Published: 15 February 2009
.... the neural tube), has a mutation in KIAA0568 . Similar phenotypes are seen in mice and in human syndromes with mutations in genes that encode centrosomal or intraflagella transport proteins. Such mutations lead to defects in primary cilia, sites where Hh signalling occurs. Here, we show that cells of talpid...
Includes: Supplementary data
Journal Articles
Journal: Development
Development (2007) 134 (11): 2159–2169.
Published: 1 June 2007
...Eiki Koyama; Blanche Young; Motohiko Nagayama; Yoshihiro Shibukawa; Motomi Enomoto-Iwamoto; Masahiro Iwamoto; Yukiko Maeda; Beate Lanske; Buer Song; Rosa Serra; Maurizio Pacifici The motor protein Kif3a and primary cilia regulate important developmental processes, but their roles in skeletogenesis...