FSTL1

Cilia effects upon perturbation of FSTL1

Cilia number / % ciliated:

Decrease

Loss-of-function effect:

Shorter cilia

Ciliogenesis screen results (4 screens)

• Kim2016: Not Reported
• 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

Phenotypes

Mouse phenotype:

increased circulating cholesterol level, increased lymphocyte cell number, increased circulating hdl cholesterol level, impaired glucose tolerance, decreased fasting circulating glucose level, decreased neutrophil cell number

Subcellular localization

cilia associated gene

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
• Cell migration & adhesion
• Signaling (Hedgehog, GPCRs, ion channels)

Function

Novel regulator of ciliogenesis in a non-cell autonomous manner due to its activity as a BMP4 inhibitor. BBS4 regulates the cellular trafficking of FSTL1. FSTL1 promotes primary cilia formation (fstl1a in zebrafish). Knockdown or mutation reduces ciliogenesis. BBS4 / IFT88 knockdown reduces FSTL1 levels, suggesting regulatory loop. FSTL1 promotes primary cilia formation (fstl1a in zebrafish). Knockdown or mutation reduces ciliogenesis. BBS4 / IFT88 knockdown reduces FSTL1 levels, suggesting regulatory loop. (28852127) fstl1a promotes primary cilia formation in early zebrafish embryos; participates in adipose tissue formation. fstl1a/b depletion affects adipocyte differentiation. Bbs4-cilia-Fstl1 axis in obesity.

Model organism evidence