Transcription factors SP5 and SP8 drive primary cilia formation in mammalian embryos Journal Article
| Authors: | Liang, Y.; Koche, R.; Chalamalasetty, R. B.; Stephen, D. N.; Kennedy, M. W.; Lao, Z.; Pang, Y.; Kuo, Y. Y.; Lee, M.; Lobo, F. P.; Huang, X.; Hadjantonakis, A. K.; Yamaguchi, T. P.; Anderson, K. V.; Joyner, A. L. |
| Article Title: | Transcription factors SP5 and SP8 drive primary cilia formation in mammalian embryos |
| Abstract: | <p>Although specific transcription factors (TFs) are known to regulate cell fate decisions, the degree to which they can stimulate formation of specific cell organelles is less clear. We used a multiomics comparison of the transcriptomes of ciliated and unciliated embryonic cells to identify TFs up-regulated in ciliated cells. We also used conditional genetics in mouse embryos and stem cells and found that the TFs SP5 and SP8 regulate cilia formation and gene expression. In embryos lacking Sp5 and Sp8, primary and motile cilia were shorter than normal and reduced in number across cell types, contributing to situs inversus and hydrocephalus. Moreover, expression of SP8 was sufficient to induce primary cilia in unciliated cells. This work will facilitate the study of cilia assembly using stem cell models and promote further understanding of human ciliopathies.</p> |
| Keywords: | mouse; genes; family; expression; cells; beta-catenin; interacts; ciliogenesis; factor rfx3; motile |
| Journal Title: | Science |
| Volume: | 389 |
| Issue: | 6763 |
| ISSN: | 0036-8075 |
| Publisher: | American Association for the Advancement of Science |
| Date Published: | 2025-08-28 |
| Start Page: | eadt5663 |
| Language: | English |
| ACCESSION: | WOS:001562017600027 |
| DOI: | 10.1126/science.adt5663 |
| PROVIDER: | wos |
| PUBMED: | 40875857 |
| Notes: | Article -- MSK Cancer Center Support Grant (P30 CA008748) acknowledged in PubMed -- MSK corresponding author is Alexandra Joyner --Source: Wos |
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