FOXN3

Cilia effects upon perturbation of FOXN3

Cilia number / % ciliated:

Increase

Loss-of-function effect:

Increase

Ciliogenesis screen results (2 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680

Phenotypes

Mouse phenotype:

decreased prepulse inhibition; preweaning lethality; complete penetrance

Subcellular localization

cilia associated gene, cytosol, nucleoplasm, nucleus

Function

Foxn3 is a key transcriptional repressor that may function to ensure the proper ciliary architecture of retinal neurons by preventing nonphotoreceptor neurons from adopting photoreceptor-like ciliary features and provide insights into the molecular mechanisms governing retinal development and ciliopathies (PMID: 40663603). The Foxn3 protein forms a complex with the Rfx3 protein, and a short protein motif shared by Foxj1, Foxn4, and Foxn3 is required for complex formation with Rfx3. The same shared motif is required for transcriptional repression by Foxn3, as well as for full transcriptional activation by Foxn4 with Rfx3. These results reveal a new layer of transcriptional regulation of genes required for cilia and show that Foxn3 prevents inappropriate expression of cilia genes in the developing retina(PMID: 39554069).Transcription factor, Potential regulator of ciliary gene expression, linking transcription to ciliary development or maintenance(39554069). Foxn3 CKO mice show ectopic ciliary gene expression and abnormal ciliogenesis in nonphotoreceptor retinal neurons (bipolar, amacrine cells). FOXN3 represses ciliary gene promoters including Foxj1 and Rfx family members.

Model organism evidence