DYNLL1

Cilia effects upon perturbation of DYNLL1

Cilia number / % ciliated:

Decreased cilia number

Loss-of-function effect:

No effect

Ciliogenesis screen results (2 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-3.77) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-4.75) PMID:29459680

Subcellular localization

basal body, centrosome, cilia, cytosol, nucleus

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
• Actin & cytoskeleton regulation
• Reproduction & sperm
• Viral interactions
• Protein processing & maturation
• Signaling (Hedgehog, GPCRs, ion channels)
• Cilia length regulation

Function

Control of retrograde intraflagellar trafficking of ciliary proteins through interaction with WDR34 and ciliogenesis by serving as dynein-1 subunits (25294941, 30649997).Dynll1(GT/GT) embryonic cilia exhibited shortening and bulging, highly similar to the characterised retrograde IFT phenotype of Dync2h1. Depletion of ATMIN or DYNLL1 in cultured cells recapitulated the in vivo ciliogenesis phenotypes and expression of DYNLL1 or the related DYNLL2 rescued the effects of loss of ATMIN, demonstrating that ATMIN primarily promotes ciliogenesis by regulating Dynll1 expression(PMID: 25294941). Cilia length was unaffected by the loss of DYNLL1.