Transcription factors SP5 and SP8 drive primary cilia formation in mammalian embryos

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 <i>Sp5</i> and <i>Sp8</i>, 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.

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Publication Details

Journal
Science
Published
2025-08-28
DOI
https://doi.org/10.1126/science.adt5663
Citations
4
Primary Topic
Genetic and Kidney Cyst Diseases
Type
article
Field-Weighted Citation Impact
7.27
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article

Transcription factors SP5 and SP8 drive primary cilia formation in mammalian embryos

Xiaofeng Huang, Anna‐Katerina Hadjantonakis, Zhimin Lao, Ravindra B. Chalamalasetty et al.
4 citations
Science
Genetic and Kidney Cyst Diseases
7.27
article

Transcription factors SP5 and SP8 drive primary cilia formation in mammalian embryos

Xiaofeng Huang, Anna‐Katerina Hadjantonakis, Zhimin Lao, Ravindra B. Chalamalasetty, Terry P. Yamaguchi, Alexandra L. Joyner, Francisco Pereira Lobo, Ying-Yi Kuo, Richard P. Koche, Yinwen Liang, Kathryn V. Anderson, Moonsup Lee, Mark W. Kennedy, Yunong Pang, Daniel N. Stephen
article en
4 citations

Abstract

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 <i>Sp5</i> and <i>Sp8</i>, 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.

ScienceVol. 389(6763)
Memorial Sloan Kettering Cancer Center (US), Cornell University (US), Frederick National Laboratory for Cancer Research (US), Center for Cancer Research (US), Weill Cornell Medicine (US)
Openalex Percentile: Top 3%
Genetic and Kidney Cyst Diseases
7.27
DOISourceOpenAlex
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