SAS-1 Is a C2 Domain Protein Critical for Centriole Integrity in C. elegans: e1004777

Centrioles are microtubule-based organelles important for the formation of cilia, flagella and centrosomes. Despite progress in understanding the underlying assembly mechanisms, how centriole integrity is ensured is incompletely understood, including in sperm cells, where such integrity is particula...

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Veröffentlicht in:PLoS genetics 2014-11, Vol.10 (11)
Hauptverfasser: Tobel, Lukas von, Mikeladze-Dvali, Tamara, Delattre, Marie, Balestra, Fernando R, Blanchoud, Simon, Finger, Susanne, Knott, Graham, Müller-Reichert, Thomas, Gönczy, Pierre
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container_issue 11
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container_title PLoS genetics
container_volume 10
creator Tobel, Lukas von
Mikeladze-Dvali, Tamara
Delattre, Marie
Balestra, Fernando R
Blanchoud, Simon
Finger, Susanne
Knott, Graham
Müller-Reichert, Thomas
Gönczy, Pierre
description Centrioles are microtubule-based organelles important for the formation of cilia, flagella and centrosomes. Despite progress in understanding the underlying assembly mechanisms, how centriole integrity is ensured is incompletely understood, including in sperm cells, where such integrity is particularly critical. We identified C. elegans sas-1 in a genetic screen as a locus required for bipolar spindle assembly in the early embryo. Our analysis reveals that sperm-derived sas-1 mutant centrioles lose their integrity shortly after fertilization, and that a related defect occurs when maternal sas-1 function is lacking. We establish that sas-1 encodes a C2 domain containing protein that localizes to centrioles in C. elegans, and which can bind and stabilize microtubules when expressed in human cells. Moreover, we uncover that SAS-1 is related to C2CD3, a protein required for complete centriole formation in human cells and affected in a type of oral-facial-digital (OFD) syndrome.
doi_str_mv 10.1371/journal.pgen.1004777
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subjects Cell cycle
Genes
Motion pictures
Nematodes
title SAS-1 Is a C2 Domain Protein Critical for Centriole Integrity in C. elegans: e1004777
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