The novel ciliogenesis regulator DYRK2 governs Hedgehog signaling during mouse embryogenesis
Mammalian Hedgehog (Hh) signaling plays key roles in embryogenesis and uniquely requires primary cilia. Functional analyses of several ciliogenesis-related genes led to the discovery of the developmental diseases known as ciliopathies.
Saishu Yoshida+8 more
doaj +1 more source
Rab8a and Rab8b are essential for several apical transport pathways but insufficient for ciliogenesis [PDF]
The small GTP-binding protein Rab8 is known to play an essential role in intracellular transport and cilia formation. We have previously demonstrated that Rab8a is required for localising apical markers in various organisms.
A. Harada+10 more
core +2 more sources
Primary cilia extending from mother centrioles are essential for vertebrate development and homeostasis maintenance. Centriolar coiled-coil protein 110 (CP110) has been reported to suppress ciliogenesis initiation by capping the distal ends of mother ...
Min Liu+15 more
semanticscholar +1 more source
To elucidate the role of centriolar satellites in ciliogenesis, we deleted the gene encoding the PCM1 protein, an integral component of satellites. PCM1 null human cells show marked defects in ciliogenesis, precipitated by the loss of specific proteins ...
Lei Wang+4 more
doaj +1 more source
A novel Cep120-dependent mechanism inhibits centriole maturation in quiescent cells [PDF]
The two centrioles of the centrosome in quiescent cells are inherently asymmetric structures that differ in age, morphology and function. How these asymmetric properties are established and maintained during quiescence remains unknown. Here, we show that
Betleja, Ewelina+3 more
core +2 more sources
Centrosome amplification disrupts renal development and causes cystogenesis [PDF]
International ...
Amanda Knoten+79 more
core +5 more sources
Bardet-Biedl syndrome (BBS) is a ciliopathy characterized by retinal dystrophy, renal cysts, obesity and polydactyly. BBS genes have been implicated in ciliogenesis, hedgehog signaling and retinal pigment epithelium maturation.
C. A. B. Hey+6 more
semanticscholar +1 more source
Combined exome and whole-genome sequencing identifies mutations in ARMC4 as a cause of primary ciliary dyskinesia with defects in the outer dynein arm [PDF]
Primary ciliary dyskinesia (PCD) is a rare, genetically heterogeneous ciliopathy disorder affecting cilia and sperm motility. A range of ultrastructural defects of the axoneme underlie the disease, which is characterised by chronic respiratory symptoms ...
Alexandros Onoufriadis+45 more
core +2 more sources
Robust interaction of IFT70 with IFT52–IFT88 in the IFT-B complex is required for ciliogenesis
In the intraflagellar transport (IFT) machinery, the IFT-B and IFT-A complexes mediate anterograde and retrograde ciliary protein trafficking, respectively.
Ryota Takei+2 more
doaj +1 more source
Gli2a protein localization reveals a role for Iguana/DZIP1 in primary ciliogenesis and a dependence of Hedgehog signal transduction on primary cilia in the zebrafish [PDF]
Background: In mammalian cells, the integrity of the primary cilium is critical for proper regulation of the Hedgehog (Hh) signal transduction pathway.
Ingham, P.W.+3 more
core +3 more sources