In embryos of most animal species, the zygotic centrosome is assembled by the centriole derived from the sperm cell and pericentriolar proteins present in the oocyte. This zygotic centrosome acts as a microtubule organizing center (MTOC) to assemble the sperm aster and mitotic spindle. As MTOC formation has been studied mainly in adult cells, very little is known about the formation of the zygotic MTOC. Here, we show that zebrafish (Danio rerio) embryos lacking either maternal or paternal Cfap53, a centriolar satellite protein, arrest during the first cell cycle. Although Cfap53 is dispensable for sperm aster function, it aids proper formation of the mitotic spindle. During cell division, Cfap53 colocalizes with γ-tubulin and with other centrosomal and centriolar satellite proteins at the MTOC. Furthermore, we find that γ-tubulin localization at the MTOC is impaired in the absence of Cfap53. Based on these results, we propose a model in which Cfap53 deposited in the oocyte and the sperm participates in the organization of the zygotic MTOC to allow mitotic spindle formation.
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http://dx.doi.org/10.1242/dev.198762 | DOI Listing |
Proc Natl Acad Sci U S A
December 2024
Department of Cell Biology, University of Texas Southwestern Medical Center, Dallas, TX 75390.
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December 2024
Laboratory of Cell Cycle Regulation, Centre for DNA Fingerprinting and Diagnostics (CDFD), Hyderabad 500039, India.
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View Article and Find Full Text PDFHGG Adv
January 2025
Institute of Cell Biology and Neurobiology, Charite - Universitatsmedizin Berlin, Berlin, Germany; Department of Pediatric Neurology, Charité - Universitatsmedizin Berlin, Berlin, Germany; Center for Chronically Sick Children, Charité - Universitatsmedizin Berlin, Berlin, Germany; German Epilepsy Center for Children and Adolescents, Charité - Universitatsmedizin Berlin, Berlin, Germany. Electronic address:
FASEB J
September 2024
Department of Gastroenterology, The First Affiliated Hospital of Kunming Medical University, Kunming, China.
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View Article and Find Full Text PDFPrimary ciliary dyskinesia (PCD) is a hereditary disease caused by genes related to motile cilia. We report two male pediatric cases of PCD caused by hemizygous pathogenic variants in the OFD1 centriole and centriolar satellite protein () gene. The variants were NM_003611.
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