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We used a multi-omics comparison of the transcriptomes of ciliated and non-ciliated embryonic cells to identify transcription factors upregulated in ciliated cells, and conditional genetics in mouse embryos and stem cells to demonstrate that SP5\/8 regulate cilia formation and gene expression. In\n                  <jats:italic>Sp5\/8<\/jats:italic>\n                  mutant embryos primary and motile cilia are shorter than normal and reduced in number across cell types, contributing to situs inversus and hydrocephalus. Moreover, expression of SP8 is sufficient to induce primary cilia in unciliated cells. This work opens new avenues for studying cilia assembly using stem cell models and offers new insights into human ciliopathies.\n                <\/jats:p>","DOI":"10.1101\/2025.06.03.657415","type":"posted-content","created":{"date-parts":[[2025,6,4]],"date-time":"2025-06-04T05:25:20Z","timestamp":1749014720000},"source":"Crossref","is-referenced-by-count":0,"title":["Transcription factors SP5 and SP8 drive primary cilia formation"],"prefix":"10.64898","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9945-3768","authenticated-orcid":false,"given":"Yinwen","family":"Liang","sequence":"first","affiliation":[{"name":"Developmental Biology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6820-5083","authenticated-orcid":false,"given":"Richard","family":"Koche","sequence":"additional","affiliation":[{"name":"Center for Epigenetics Research, Memorial 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