Drosophila Alms1 proteins regulate centriolar cartwheel assembly by enabling Plk4-Ana2 amplification loop
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ABSTRACT: Centrioles play a central role in cell division by recruiting pericentriolar material (PCM) to form the centrosome. Alterations in centriole number or function can lead to various diseases, including cancer or microcephaly. Centriole duplication is a highly conserved process in eukaryotes. Here, we show that the two Drosophila orthologs of the Alström syndrome protein 1 (Alms1a and Alms1b) cooperate to regulate centriole duplication in various fly tissues. Using ultrastructure expansion microscopy (UExM), we reveal that Alms1a is a PCM protein that is loaded proximally on centrioles at the onset of procentriole formation, whereas Alms1b caps the base of mature centrioles. We demonstrate that chronic loss of Alms1 proteins (null-RNA allele) affects PCM maturation, whereas their acute loss (RNAi knockdown) completely disrupts procentriole formation before Sas-6 cartwheel assembly. We further show that Alms1 proteins are required for the amplification of the Plk4-Ana2 pool at the duplication site and the subsequent Sas-6 recruitment. Thus, our findings establish that Alms1 proteins are critical, but highly buffered, regulators of PCM and cartwheel assembly in flies.
SUBMITTER: Marine Brunet
PROVIDER: S-SCDT-10_1038-S44318-025-00382-8 | biostudies-other |
REPOSITORIES: biostudies-other
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