DIDO1

Cilia effects upon perturbation of DIDO1

Cilia number / % ciliated:

No effect

Loss-of-function effect:

Longer cilia

Overexpression effect:

Shorter cilia

Ciliogenesis screen results (3 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
• Pusapati et al. 2018 (CRISPR) [CRISPR]: Negative Regulator (Hh signaling, pos_rank=589, lfc=-0.25) PMID:30270045

Subcellular localization

basal body, nucleus

Functional category

• Ciliary assembly/disassembly

Function

As the same organelle that sprouts the cilium in interphase organizes the mitotic spindle during cell division, cilia must be shortened and disassembled before mitosis11. The subcellular distribution and recruitment of Dido3 to spindle poles hint at a role for the protein in this process. To determine whether Dido3 has a role in the regulation of cilium size, we measured the primary cilium length in serum-starved WT and ΔNT MEF (Fig. 2). Labelling with antibodies against the proliferation marker proliferating-cell nuclear antigen confirmed cell cycle arrest after serum starvation (Supplementary Fig. 3). Although cell cycle arrest readily produced cilia in the WT and ΔNT MEFs (Fig. 2a), average cilium size was significantly reduced in the ΔNT MEF (Fig. 2b). As the independent marker ARL13B34 distributed along, but did not extend significantly beyond the Ac-tub signal (Fig. 2c,d), Ac-tub accurately determined cilium size for WT and ΔNT MEF. Subsequent quantification revealed that cilium size negatively correlated with Dido3/Dido3ΔNT protein levels at the basal body (Supplementary Fig. 4). As Dido3ΔNT is more efficiently recruited to the basal body than WT Dido3 (Fig. 1), these data suggest that Dido3 negatively regulates cilium size(24667272).