C. elegans SAS-1 ensures centriole integrity and ciliary function, and operates with SSNA-1
Centrioles are microtubule-based organelles critical for signaling, motility and division. The microtubule-binding protein SAS-1 is homologous to the human ciliopathy component C2CD3 and contributes to centriole integrity in C. elegans, but how this function is exerted is incompletely understood. Here, through the generation of a null allele and analysis with U-Ex-STED, we establish that SAS-1 is dispensable for the onset of centriole assembly, but essential for organelle integrity during oogenesis, spermatogenesis and in the early embryo. Additionally, we uncover that SAS-1 is present at the transition zone of sensory neurons, and contributes in a partially redundant manner to ciliary function. Furthermore, we investigate the relationship between SAS-1 and the C. elegans Sjögren's Syndrome Nuclear Antigen 1 protein SSNA-1, establishing that SSNA-1 localizes next to the SAS-1 C-terminus in the centriole architecture. Moreover, through molecular epistasis experiments with null alleles of both components, we reveal that SAS-1 is essential for SSNA-1 localization to centrioles during oogenesis and to the transition zone during ciliogenesis. Moreover, using a heterologous human cell assay, we establish that SAS-1 recruits SSNA-1 to microtubules. Overall, our findings help clarify how SAS-1, together with SSNA-1, ensures centriole integrity, and reveal that it contributes to cilium function.
Authors
- Pierre Gönczy (ORCID: https://orcid.org/0000-0002-6305-6883)
- Coralie Busso (ORCID: https://orcid.org/0009-0003-4081-9416)
- Alexander Woglar (ORCID: https://orcid.org/0009-0008-5304-2497)
- Georgios N. Hatzopoulos (ORCID: https://orcid.org/0000-0003-4382-4459)
- Tatiana Favez
- Keshav Jha (ORCID: https://orcid.org/0000-0003-2482-6723)
Institutions
- École Polytechnique Fédérale de Lausanne (CH)
Publication Details
- Journal
- PLoS Genetics
- Published
- 2025-10-22
- DOI
- https://doi.org/10.1371/journal.pgen.1011912
- Citations
- 3
- Primary Topic
- Genetic and Kidney Cyst Diseases
- Type
- article
- Field-Weighted Citation Impact
- 5.45