FOXN4

Cilia effects upon perturbation of FOXN4

Loss-of-function effect:

Defective ciliogenesis

Ciliogenesis screen results (5 screens)

• Kim2016: No effect
• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-5.54) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
• Pusapati et al. 2018 (CRISPR) [CRISPR]: Negative Regulator (Hh signaling, pos_rank=104, lfc=0.76) PMID:30270045

Phenotypes

Mouse phenotype:

increased total body fat amount, no spontaneous movement, decreased prepulse inhibition, preweaning lethality, incomplete penetrance, abnormal spleen morphology, decreased lean body mass, abnormal tooth color, abnormal skin morphology, increased cd4-positive, cd25-positive, alpha-beta regulatory t cell number

Subcellular localization

basal body, nucleus

References: PMID:27864379

Functional category

• Ciliary assembly/disassembly
• Muscle contraction & physiology

Function

Downstream target of Multicilin required for multiciliated cells differentiation (MCC). Complements Foxj1 transcriptionally by co- activating Foxj1 target genes. Ensures robust gene expression during MCC differentiation, allowing large numbers of specialized organelles to be assembled. When Foxn4 activity is inhibited in Xenopus embryos, MCCs show transient ciliogenesis defects similar to those seen in mutants of Foxj1. Foxn4 promotes gene expression required for the formation of multiple motile cilia (27864379)