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. 2014 May 22;111(23):8541–8546. doi: 10.1073/pnas.1323161111

Fig. 6.

Fig. 6.

The ICK mutation identified in human ECO syndrome causes defects in ciliogenesis and HH signaling. ICK-3FLAG mutant (R272Q and R272A) plasmids were transfected into cultured cells. After transfection, cells were stained with fluorescent Arl13b (green) and FLAG (red) antibodies. (A and B) Ciliary length was increased by approximately twofold by overexpression of ICK mutant proteins. Transfected ICK mutant proteins were localized at the bulged tips of primary cilia. (Scale bars, 5 µm.) (C) SHH responsiveness was severely compromised by overexpression of wild-type or mutant forms of ICK in cultured cells treated with SHHN-conditioned media. Error bars in B and C represent SEM. *P < 0.01; **P < 0.05 (Student’s t tests).