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The SASS6(I62T) mutation corresponds to SAS-6(L69T) in Caenorhabditis elegans. Given that SAS-6 is highly conserved, we modeled this mutation in C. elegans and examined the sas-6(L69T) effect on centrosome duplication, ciliogenesis, and dendrite morphogenesis. Our studies revealed that all the above processes are perturbed by the sas-6(L69T) mutation. Specifically, C. elegans carrying the sas-6(L69T) mutation exhibit an increased failure of centrosome duplication in a sensitized genetic background. Further, worms carrying this mutation also display shortened phasmid cilia, an abnormal phasmid cilia morphology, shorter phasmid dendrites, and chemotaxis defects. Our data show that the centrosome duplication defects caused by this mutation are only uncovered in a sensitized genetic background, indicating that these defects are mild. However, the ciliogenesis and dendritic defects caused by this mutation are evident in an otherwise wild-type background, indicating that they are stronger defects. Thus, our studies shed light on the novel mechanisms by which the sas-6(L69T) mutation could contribute to the incidence of primary microcephaly in humans.<\/jats:p>","DOI":"10.1093\/genetics\/iyad105","type":"journal-article","created":{"date-parts":[[2023,6,9]],"date-time":"2023-06-09T09:29:48Z","timestamp":1686302988000},"source":"Crossref","is-referenced-by-count":13,"title":["A primary microcephaly-associated\n                    <i>sas-6<\/i>\n                    mutation perturbs centrosome duplication, dendrite morphogenesis, and ciliogenesis in\n                    <i>Caenorhabditis elegans<\/i>"],"prefix":"10.1093","volume":"224","author":[{"given":"Mary","family":"Bergwell","sequence":"first","affiliation":[{"name":"Oklahoma Medical Research Foundation , Cell Cycle & Cancer Biology Research Program, Oklahoma City, OK 73104 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amy","family":"Smith","sequence":"additional","affiliation":[{"name":"Pfizer Inc. , Pharmaceutical R&D \u2013 Drug Product Design & Development, Chesterfield, MO 63017 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ellie","family":"Smith","sequence":"additional","affiliation":[{"name":"University of Tulsa , Department of Chemistry and Biochemistry, Tulsa, OK 74104 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carter","family":"Dierlam","sequence":"additional","affiliation":[{"name":"University of Tulsa , Department of Chemistry and Biochemistry, Tulsa, OK 74104 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ramon","family":"Duran","sequence":"additional","affiliation":[{"name":"University of Tulsa , Department of Chemistry and Biochemistry, Tulsa, OK 74104 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erin","family":"Haastrup","sequence":"additional","affiliation":[{"name":"University of Tulsa , Department of Chemistry and Biochemistry, Tulsa, OK 74104 , 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