ArticleProceedings of the National Academy of Sciences of the United States of America2014
Conserved TCP domain of Sas-4/CPAP is essential for pericentriolar material tethering during centrosome biogenesis.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 49 papers.
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49 citing papers in PubMed, 69 citations in OpenAlex.
- Unraveling the molecular details of centriole elongation.Trends open · 2026Article
- Microcephaly-associated genes asp and Sas4 influence chromatin organization and nuclear lamina structure in Drosophila melanogaster.Development (Cambridge, England) · 2026Article
- Cytoplasmic control of spindle architecture.Nature cell biology · 2025Article
- Cell state-specific cytoplasmic density controls spindle architecture and scaling.Nature cell biology · 2025Article
- Expanding the mutational spectrum of congenital microcephaly in Pakistani families.Frontiers in genetics · 2025Article
- Brain organoids: A new tool for modelling of neurodevelopmental disorders.Journal of cellular and molecular medicine · 2024Review
- Inhibiting centrosome clustering reduces cystogenesis and improves kidney function in autosomal dominant polycystic kidney disease.JCI insight · 2024Article
- Structural insights into the mechanism of GTP initiation of microtubule assembly.Nature communications · 2023Article
- CPAP insufficiency leads to incomplete centrioles that duplicate but fragment.The Journal of cell biology · 2022Article
- Review
- Modeling Human Primary Microcephaly With hiPSC-Derived Brain Organoids Carrying CPAP-E1235V Disease-Associated Mutant Protein.Frontiers in cell and developmental biology · 2022Article
- Time is of the essence: the molecular mechanisms of primary microcephaly.Genes & development · 2021Review
- Human centrosome organization and function in interphase and mitosis.Seminars in cell & developmental biology · 2021Review
- Human pluripotent stem cell-derived brain organoids as in vitro models for studying neural disorders and cancer.Cell & bioscience · 2021Review
- Modifier Genes in Microcephaly: A Report onGenes · 2021Article
- Loss of the centrosomal protein Cenpj leads to dysfunction of the hypothalamus and obesity in mice.Science China. Life sciences · 2021Article
- ESI mutagenesis: a one-step method for introducing mutations into bacterial artificial chromosomes.Life science alliance · 2021Article
- Sas-4 Colocalizes with the Ciliary Rootlets of theJournal of developmental biology · 2021Article
- Zika Virus and Neuropathogenesis: The Unanswered Question of Which Strain Is More Prone to Causing Microcephaly and Other Neurological Defects.Frontiers in cellular neuroscience · 2021Review
- Structure-Based Deep Mining Reveals First-Time Annotations for 46 Percent of the Dark Annotation Space of the 9,671-Member Superproteome of the Nucleocytoplasmic Large DNA Viruses.Journal of virology · 2020Article
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16 authors at 6 institutions in 5 countries.
Funding
Abstract
Pericentriolar material (PCM) recruitment to centrioles forms a key step in centrosome biogenesis. Deregulation of this process leads to centrosome aberrations causing disorders, one of which is autosomal recessive primary microcephaly (MCPH), a neurodevelopmental disorder where brain size is reduced. During PCM recruitment, the conserved centrosomal protein Sas-4/CPAP/MCPH6, known to play a role in centriole formation, acts as a scaffold for cytoplasmic PCM complexes to bind and then tethers them to centrioles to form functional centrosomes. To understand Sas-4's tethering role, we determined the crystal structure of its T complex protein 10 (TCP) domain displaying a solvent-exposed single-layer of β-sheets fold. This unique feature of the TCP domain suggests that it could provide an "extended surface-like" platform to tether the Sas-4-PCM scaffold to a centriole. Functional studies in Drosophila, human cells, and human induced pluripotent stem cell-derived neural progenitor cells were used to test this hypothesis, where point mutations within the 9-10th β-strands (β9-10 mutants including a MCPH-associated mutation) perturbed PCM tethering while allowing Sas-4/CPAP to scaffold cytoplasmic PCM complexes. Specifically, the Sas-4 β9-10 mutants displayed perturbed interactions with Ana2, a centrosome duplication factor, and Bld-10, a centriole microtubule-binding protein, suggesting a role for the β9-10 surface in mediating protein-protein interactions for efficient Sas-4-PCM scaffold centriole tethering. Hence, we provide possible insights into how centrosomal protein defects result in human MCPH and how Sas-4 proteins act as a vehicle to tether PCM complexes to centrioles independent of its well-known role in centriole duplication.
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