ArticleNature communications2017
Human microcephaly protein RTTN interacts with STIL and is required to build full-length centrioles.
Article in Nature communications, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 32 papers.
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Who cites it
32 citing papers in PubMed, 47 citations in OpenAlex.
- Microtubule-Associated Proteins: From Dynamic Regulation of Microtubules to Cellular Architecture.Cells · 2026Review
- Critical constituents and assembly principles of centriole biogenesis in human cells.Nature reviews. Molecular cell biology · 2026Review
- Rewiring cancer: 3D genome determinants of cancer hallmarks.Current opinion in genetics & development · 2025Review
- Radial Microbrain (Micrencephaly) Is Caused by a Recurrent Variant in theNeurology. Genetics · 2025Article
- Predicting potential target genes in molecular biology experiments using machine learning and multifaceted data sources.iScience · 2024Article
- Impaired Reorganization of Centrosome Structure Underlies Human Infantile Dilated Cardiomyopathy.Circulation · 2023Article
- A biallelic frameshift indel in PPP1R35 as a cause of primary microcephaly.American journal of medical genetics. Part A · 2023Article
- Case Report: Novel biallelic moderately damaging variants inFrontiers in pediatrics · 2023Article
- Molecular subtypes based on centrosome-related genes can predict prognosis and therapeutic responsiveness in patients with low-grade gliomas.Frontiers in oncology · 2023Article
- A novel HIF1α-STIL-FOXM1 axis regulates tumor metastasis.Journal of biomedical science · 2022Article
- 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
- Ana1 helps recruit Polo to centrioles to promote mitotic PCM assembly and centriole elongation.Journal of cell science · 2021Article
- Human Microcephaly ProteinCells · 2021Article
- Superresolution characterization of core centriole architecture.The Journal of cell biology · 2021Article
- Review
- Tissue specific requirement of Drosophila Rcd4 for centriole duplication and ciliogenesis.The Journal of cell biology · 2020Article
- Protein phosphatase 1 regulatory subunit 35 is required for ciliogenesis, notochord morphogenesis, and cell-cycle progression during murine development.Developmental biology · 2020Article
- With Age Comes Maturity: Biochemical and Structural Transformation of a Human Centriole in the Making.Cells · 2020Review
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Mutations in many centriolar protein-encoding genes cause primary microcephaly. Using super-resolution and electron microscopy, we find that the human microcephaly protein, RTTN, is recruited to the proximal end of the procentriole at early S phase, and is located at the inner luminal walls of centrioles. Further studies demonstrate that RTTN directly interacts with STIL and acts downstream of STIL-mediated centriole assembly. CRISPR/Cas9-mediated RTTN gene knockout in p53-deficient cells induce amplification of primitive procentriole bodies that lack the distal-half centriolar proteins, POC5 and POC1B. Additional analyses show that RTTN serves as an upstream effector of CEP295, which mediates the loading of POC1B and POC5 to the distal-half centrioles. Interestingly, the naturally occurring microcephaly-associated mutant, RTTN (A578P), shows a low affinity for STIL binding and blocks centriole assembly. These findings reveal that RTTN contributes to building full-length centrioles and illuminate the molecular mechanism through which the RTTN (A578P) mutation causes primary microcephaly.Mutations in many centriolar protein-encoding genes cause primary microcephaly. Here the authors show that human microcephaly protein RTTN directly interacts with STIL and acts downstream of STIL-mediated centriole assembly, contributing to building full-length centrioles.
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