SIRT1

Cilia effects upon perturbation of SIRT1

Cilia number / % ciliated:

Decreased cilia number

Loss-of-function effect:

Longer cilia

Overexpression effect:

Decreased

Ciliogenesis screen results (4 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-4.82) 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=-4.52) PMID:41160700

Phenotypes

Mouse phenotype:

abnormal maxilla morphology, increased basophil cell number, abnormal scapula morphology, rrow eye opening, abnormal pelvic girdle bone morphology, abnormal tooth morphology, preweaning lethality, incomplete penetrance, abnormal snout morphology, abnormal sinus arrhythmia, abnormal iris morphology

Subcellular localization

cilia associated gene

Functional category

• Ciliary assembly/disassembly
• Actin & cytoskeleton regulation
• Metabolism
• T cell biology
• Reproduction & sperm
• Cell migration & adhesion
• Signaling (Hedgehog, GPCRs, ion channels)
• Cardiac & muscle development
• ECM & connective tissue
• Transcription regulation

Function

Small interfering RNA (siRNA)-mediated knockdown of SIRT1 in HuCCT1 cells induced cilia formation, whereas overexpression of SIRT1 in normal cholangiocytes suppressed ciliary expression(PMID: 34322899).

Model organism evidence