FOXJ1

forkhead box J1

Ensembl:
ENSG00000129654
UniProt:
Q92949
OMIM:
602291
Synonyms:
FKHL13, HFH-4, HFH4

Cilia effects upon perturbation of FOXJ1

Loss-of-function effect:
Shorter cilia
Overexpression effect:
Longer

Ciliogenesis screen results (5 screens)

  • Kim2016: Not Reported
  • Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-2.76) 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=697, lfc=1.02) PMID:30270045

Ciliopathy associations

  • Hydrocephalus
  • Primary Ciliary Dyskinesia

Subcellular localization

cilia associated gene, nucleus

Functional category

  • Ciliary assembly/disassembly
  • Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
  • Actin & cytoskeleton regulation
  • T cell biology
  • Protein processing & maturation
  • Cell migration & adhesion
  • Transcription regulation
  • Cilia length regulation

Function

Transcription factor that controls motile cilia formation ( 19011630). Activates the transcription of numerous known ciliary genes and specifically regulates motile cilia biogenesis ( 30881373). Regulation of programs promoting basal body docking and axoneme formation. Role of FOXJ1 in the differentiation of motile ciliated cells is modulated by the transcription factor Not homeobox (NOTO) and the regulatory factor X (RFX)- family ( 23822649) Transcription factor that controls motile cilia formation ( 19011630). Activates the transcription of numerous known ciliary genes and specifically regulates motile cilia biogenesis ( 30881373). Regulation of programs promoting basal body docking and axoneme formation. Role of FOXJ1 in the differentiation of motile ciliated cells is modulated by the transcription factor Not homeobox (NOTO) and the regulatory factor X (RFX)- family ( 23822649) In the presence of CSE … the average cilia length was significantly higher in Lenti-FOXJ1–infected cells (5.2 μm) than in Lenti-control–infected cells. (24828273) Suppressed Foxj1 expression has led to shorter cilia length in cilia cell cultures, whereas overexpression of exogenous Foxj1 reversed the effect. The level of Foxj1 was positively correlated to the airway cilia length, as patients with lower Foxj1 expression showed shorter cilia length(36929635)

Model organism evidence

Mus musculus (1 reference)

Foxn3 is part of a transcriptional network that regulates primary cilia in the developing retina.

PMIDs: 41766387