Dilatory is a <i>Drosophila</i> protein related to AZI1 (CEP131) that is located at the ciliary base and required for cilium formation

Ma, Lina; Jarman, Andrew P.

doi:10.1242/jcs.084798

Cited by 67 publications

(96 citation statements)

References 49 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Proteins that, putatively, interacted with Ccdc13 with the greatest peptide coverage were PCM1, pericentrin, Cep290, Cep215, Cep131, Cep90 and Cep72 (Fig. 3A), which have all been previously shown to localise to centriolar satellites (Balczon et al, 1994;Dammermann and Merdes, 2002;Kim et al, 2008;Kim et al, 2012;Lee and Rhee, 2010;Lopes et al, 2011;Ma and Jarman, 2011;Stowe et al, 2012). Additionally, we confirmed that YFP-Ccdc13 colocalised with PCM1 and pericentrin (Fig.…”

Section: Ccdc13 Is a Novel Human Centriolar Satellite Proteinsupporting

confidence: 81%

Ccdc13; a novel human centriolar satellite protein required for ciliogenesis and genome stability

Staples

Myers

Beveridge

et al. 2014

Journal of Cell Science

View full text Add to dashboard Cite

Here, we identify coiled-coil domain-containing protein 13 (Ccdc13) in a genome-wide RNA interference screen for regulators of genome stability. We establish that Ccdc13 is a newly identified centriolar satellite protein that interacts with PCM1, Cep290 and pericentrin and prevents the accumulation of DNA damage during mitotic transit. Depletion of Ccdc13 results in the loss of microtubule organisation in a manner similar to PCM1 and Cep290 depletion, although Ccdc13 is not required for satellite integrity. We show that microtubule regrowth is enhanced in Ccdc13-depleted cells, but slowed in cells that overexpress Ccdc13. Furthermore, in serumstarved cells, Ccdc13 localises to the basal body, is required for primary cilia formation and promotes the localisation of the ciliopathy protein BBS4 to both centriolar satellites and cilia. These data highlight the emerging link between DNA damage response factors, centriolar and peri-centriolar satellites and ciliaassociated proteins and implicate Ccdc13 as a centriolar satellite protein that functions to promote both genome stability and cilia formation.

show abstract

Section: Ccdc13 Is a Novel Human Centriolar Satellite Proteinsupporting

confidence: 81%

Ccdc13; a novel human centriolar satellite protein required for ciliogenesis and genome stability

Staples

Myers

Beveridge

et al. 2014

Journal of Cell Science

View full text Add to dashboard Cite

show abstract

“…As part of this program, ATO activates the expression of both fd3F and Rfx in order to generate the motile mechanosensory cilia that extend from these neurons (Cachero et al, 2011). Interestingly, ATO has also been shown to activate directly the expression of a ciliary component, Dilatory/CEP131, indicating that ATO can contribute to ciliogenesis independent of the RFX/FD3F cassette (Cachero et al, 2011;Ma and Jarman, 2011). Indeed, this function of ATO may represent a fundamental mechanism for generating mechanosensory cilia because, in zebrafish, an ATO ortholog (ATOH1B) activates a foxj1 paralog in the hair cells of the inner ear, leading to the formation of the immotile kinocilia (Yu et al, 2011).…”

Section: Cell Type-specific Transcription Factors That Regulate Ciliamentioning

confidence: 99%

Switching on cilia: transcriptional networks regulating ciliogenesis

et al. 2014

View full text Add to dashboard Cite

Cilia play many essential roles in fluid transport and cellular locomotion, and as sensory hubs for a variety of signal transduction pathways. Despite having a conserved basic morphology, cilia vary extensively in their shapes and sizes, ultrastructural details, numbers per cell, motility patterns and sensory capabilities. Emerging evidence indicates that this diversity, which is intimately linked to the different functions that cilia perform, is in large part programmed at the transcriptional level. Here, we review our understanding of the transcriptional control of ciliary biogenesis, highlighting the activities of FOXJ1 and the RFX family of transcriptional regulators. In addition, we examine how a number of signaling pathways, and lineage and cell fate determinants can induce and modulate ciliogenic programs to bring about the differentiation of distinct cilia types.

show abstract

“…Little is known about Cep131's cellular function, although Zebrafish and Drosophila lacking Cep131 orthologues have shortened cilia, and display phenotypes reminiscent of human ciliopathies such as Bardet-Biedl syndrome (Ma and Jarman, 2011;Wilkinson et al, 2009). Interestingly, Cep131 depletion results in reduced ciliogenesis in human epithelial cells, with those cilia that do form exhibiting gross morphological defects (Graser et al, 2007).…”

Section: Introductionmentioning

confidence: 99%

The centriolar satellite protein Cep131 is important for genome stability.

Staples

Myers

Beveridge

et al. 2012

Journal of Cell Science

View full text Add to dashboard Cite

SummaryThe centrosome acts as a centre for microtubule organisation and plays crucial roles in cell polarity, migration, growth and division. Cep131 has recently been described as a basal body component essential for cilium formation, but its function in non-ciliogenic cells is unknown. We identified human Cep131 (also known as AZI1) in a screen for regulators of genome stability. We show that centrosomal localisation of Cep131 is cell-cycle-regulated and requires both an intact microtubule network and a functional dynein-dynactin transport system. Cep131 is recruited to centriolar satellites by PCM1, and localised to the centriolar core region by both pericentrin and Cep290. Depletion of Cep131 results in a reduction in proliferation rate, centriole amplification, an increased frequency of multipolar mitosis, chromosomal instability and an increase in post-mitotic DNA damage. These data therefore highlight the importance of human Cep131 for maintaining genomic integrity.

show abstract

Dilatory is a Drosophila protein related to AZI1 (CEP131) that is located at the ciliary base and required for cilium formation

Cited by 67 publications

References 49 publications

Ccdc13; a novel human centriolar satellite protein required for ciliogenesis and genome stability

Ccdc13; a novel human centriolar satellite protein required for ciliogenesis and genome stability

Switching on cilia: transcriptional networks regulating ciliogenesis

The centriolar satellite protein Cep131 is important for genome stability.

Contact Info

Product

Resources

About