NOTCH1

Cilia effects upon perturbation of NOTCH1

Cilia number / % ciliated:

Unknown

Loss-of-function effect:

Shorter cilia

Overexpression effect:

Unknown

Ciliogenesis screen results (4 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
• Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-3.22) PMID:41160700

Phenotypes

Mouse phenotype:

abnormal vertebrae morphology, increased grip strength, increased startle reflex, preweaning lethality, complete penetrance; abnormal vertebrae morphology; increased grip strength; increased startle reflex; preweaning lethality; complete penetrance

Ciliopathy associations

• Caroli Disease

Subcellular localization

basal body, cilia, cilia associated gene, cytosol, endosome, nucleoplasm, nucleus

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
• Actin & cytoskeleton regulation
• Neurogenesis & migration
• Viral interactions
• Cell migration & adhesion
• Signaling (Hedgehog, GPCRs, ion channels)
• Cardiac & muscle development
• Axon guidance & growth
• ECM & connective tissue
• Transcription regulation; Motile cilium & axoneme

Function

It is glycosylated by GALNT11 to establish an essential balance between motile and immotile cilia at the left-right organiser, controlling laterality (24226769).; Notch1 activation was most probably induced by Foxj1-expressing cells, which suggests that Notch1-mediated lateral inhibition regulates the selection of Clara cells at the expense of ciliated cells(PMID: 20048339).