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000267357 1001_ $$aRayamajhi, Dheeraj$$b0
000267357 245__ $$aThe forkhead transcription factor Foxj1 controls vertebrate olfactory cilia biogenesis and sensory neuron differentiation.
000267357 260__ $$aLawrence, KS$$bPLoS$$c2024
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000267357 520__ $$aIn vertebrates, olfactory receptors localize on multiple cilia elaborated on dendritic knobs of olfactory sensory neurons (OSNs). Although olfactory cilia dysfunction can cause anosmia, how their differentiation is programmed at the transcriptional level has remained largely unexplored. We discovered in zebrafish and mice that Foxj1, a forkhead domain-containing transcription factor traditionally linked with motile cilia biogenesis, is expressed in OSNs and required for olfactory epithelium (OE) formation. In keeping with the immotile nature of olfactory cilia, we observed that ciliary motility genes are repressed in zebrafish, mouse, and human OSNs. Strikingly, we also found that besides ciliogenesis, Foxj1 controls the differentiation of the OSNs themselves by regulating their cell type-specific gene expression, such as that of olfactory marker protein (omp) involved in odor-evoked signal transduction. In line with this, response to bile acids, odors detected by OMP-positive OSNs, was significantly diminished in foxj1 mutant zebrafish. Taken together, our findings establish how the canonical Foxj1-mediated motile ciliogenic transcriptional program has been repurposed for the biogenesis of immotile olfactory cilia, as well as for the development of the OSNs.
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000267357 650_7 $$2NLM Chemicals$$aForkhead Transcription Factors
000267357 650_2 $$2MeSH$$aAnimals
000267357 650_2 $$2MeSH$$aHumans
000267357 650_2 $$2MeSH$$aMice
000267357 650_2 $$2MeSH$$aZebrafish: genetics
000267357 650_2 $$2MeSH$$aZebrafish: metabolism
000267357 650_2 $$2MeSH$$aCilia: metabolism
000267357 650_2 $$2MeSH$$aForkhead Transcription Factors: genetics
000267357 650_2 $$2MeSH$$aForkhead Transcription Factors: metabolism
000267357 650_2 $$2MeSH$$aOlfactory Receptor Neurons
000267357 650_2 $$2MeSH$$aOlfactory Mucosa
000267357 7001_ $$aEge, Mert$$b1
000267357 7001_ $$aUkhanov, Kirill$$b2
000267357 7001_ $$aRingers, Christa$$b3
000267357 7001_ $$aZhang, Yiliu$$b4
000267357 7001_ $$aJung, Inyoung$$b5
000267357 7001_ $$aD'Gama, Percival P$$b6
000267357 7001_ $$aLi, Summer Shijia$$b7
000267357 7001_ $$0P:(DE-2719)2811286$$aCosacak, Mehmet Ilyas$$b8$$udzne
000267357 7001_ $$0P:(DE-2719)2811030$$aKizil, Caghan$$b9$$udzne
000267357 7001_ $$aPark, Hae-Chul$$b10
000267357 7001_ $$aYaksi, Emre$$b11
000267357 7001_ $$aMartens, Jeffrey R$$b12
000267357 7001_ $$aBrody, Steven L$$b13
000267357 7001_ $$aJurisch-Yaksi, Nathalie$$b14
000267357 7001_ $$00000-0002-6636-1429$$aRoy, Sudipto$$b15
000267357 773__ $$0PERI:(DE-600)2126773-X$$a10.1371/journal.pbio.3002468$$gVol. 22, no. 1, p. e3002468 -$$n1$$pe3002468$$tPLoS biology$$v22$$x1544-9173$$y2024
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