FGFR1OP2

Cilia effects upon perturbation of FGFR1OP2

Cilia number / % ciliated:

Decrease

Loss-of-function effect:

Shorter cilia

Ciliogenesis screen results (5 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
• Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-2.63) PMID:41160700

Phenotypes

Mouse phenotype:

abnormal placenta vasculature, cleft palate, abnormal limb morphology, abnormal tail morphology, abnormal placenta size, abnormal blood vessel morphology, preweaning lethality, complete penetrance, abnormal placenta morphology, embryonic growth retardation, abnormal craniofacial morphology, abnormal skin morphology, edema

Mouse ciliopathy phenotype:

microphthalmia, abnormal vitreous body morphology, cataract, abnormal reti morphology, persistence of hyaloid vascular system

Subcellular localization

cilia associated gene, cytosol

Functional category

• Ciliary assembly/disassembly
• Signaling (Hedgehog, GPCRs, ion channels)

Function

SLMAP3 modulates PCP at the level of primary cilium, a function that might be shared with its partner FGFR1OP2, with mechanisms involving protein stability of DVL3 and STRIPAK independent of Hippo sig lling ( 39417621). FGFR1OP2-deficient MEFs show significant changes in ciliated cell number and primary cilia length. STRIPAK complex member; functions alongside SLMAP3 in ciliogenesis.

Model organism evidence