ATG16L1

Cilia effects upon perturbation of ATG16L1

Ciliogenesis screen results (5 screens)

• Kim2016: Not Reported
• Wheway et al. 2015 (siRNA) [siRNA]: Hyper-ciliogenesis (z=2.60) 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=-2.50) PMID:41160700

Phenotypes

Mouse phenotype:

preweaning lethality, complete penetrance

Subcellular localization

cilia

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
• Actin & cytoskeleton regulation
• Reproduction & sperm
• Viral interactions

Function

ATG16L1 regulates ciliogenesis independently of macro-autophagy. ATG16L1 interacts with ciliary protein IFT20 via WD40 domain to regulate ciliogenesis. ATG16L1 interacts with the phosphoinositide-associated phosphatase INPP5E. Loss of ATG16L1 interaction with INPP5E leads to a default of PI4P at the primary cilium (33910006). Loss of ATG16L1 results in abnormal cilium structures, especially in phosphoinositide pattern and membrane composition, and mistrafficking of INPP5E(PMID: 34057021).