OSBPL3

Cilia effects upon perturbation of OSBPL3

Ciliogenesis screen results (4 screens)

• Kim2016: Not Reported
• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-3.33) 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 lean body mass; decreased total body fat amount; increased bone mineral density; increased bone mineral content

Subcellular localization

cilia associated gene, cytosol, nucleoli

Function

ORP3 protein as a lipid transfer protein for the ciliary cholesterol trafficking from peroxisomes to primary cilia (Fig 5). Many ORP3‐positive punctate signals were detected not only at the ciliary pocket, but also at other cellular regions (Fig 5E and H). In contrast, the FRB‐FKBP system‐mediated peroxisome targeting to the ciliary pocket did not restore the ciliary cholesterol insufficiency in ORP3 −/− hTERT‐RPE1 cells (Fig 8A and B), suggesting that ORP3 is required for at least the peroxisome‐mediated cholesterol trafficking into the ciliary membrane (PMID: 28122914).