BICC1

Cilia effects upon perturbation of BICC1

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=-2.71) PMID:41160700

Phenotypes

Mouse phenotype:

preweaning lethality, complete penetrance, abnormal heart looping, preweaning lethality, incomplete penetrance, abnormal blood vessel morphology, decreased locomotor activity, abnormal placenta morphology, edema, abnormal placenta vasculature

Mouse ciliopathy phenotype:

abnormal heart morphology, abnormal embryo turning, small kidney, abnormal kidney morphology

Human ciliopathy phenotype:

multicystic dysplastic kidney

Ciliopathy associations

• Cystic Kidney Disease
• Cystic Renal Dysplasia

Subcellular localization

cilia

Functional category

• Ciliary assembly/disassembly

Function

Localization of BICC1 in cilia is controversial. Targeted i ctivation of BicC randomizes left-right (LR) asymmetry by disrupting the pla r alignment of motile cilia required for cilia-driven fluid flow. Depending on its SAM domain, BicC can uncouple Dvl2 sig ling from the canonical Wnt pathway. The SAM domain concentrates BicC in cytoplasmic structures harboring R -processing bodies (P-bodies) and Dvl2. BicC links the orientation of cilia with PCP, possibly by regulating R silencing in P-bodies (19666828).