Abstract
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.
Highlights
Centrioles are small microtubule-based organelles that are critical for the formation of cilia and flagella across eukaryotic evolution, as well as for that of centrosomes in animal cells
We identified sas-1 as a component that contributes to centriole stability in C. elegans
Centrioles that lack sas-1 function loose their integrity, and our analysis reveals that sas-1 is important for sperm-derived centrioles
Summary
Centrioles are small microtubule-based organelles that are critical for the formation of cilia and flagella across eukaryotic evolution, as well as for that of centrosomes in animal cells. Centriolar microtubules exhibit unusual stability, which is thought to contribute to the integrity of the entire organelle That centrioles retain such integrity is probably key to withstand mechanical stresses generated by cilia, flagella and centrosomes, and to ensure proper assembly of new centrioles in proliferating cells. Homologs of the core components initially identified in worms turned out to be critical for centriole formation from algae to humans [16,17]. This indicates that C. elegans can serve as a model to discover fundamental and conserved features of centriole biology
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