Figure 6.

IFT-B proteins in INPP5E^D477N/D477N organoids

Control and INPP5E^D477N/D477N organoids were immunostained with the indicated markers.

(A–P) IFT81 and IFT88 were expressed in a higher proportion of ciliary axonemes and at increased levels.

(Q–V) UB expression increased at the base, but not in the axoneme, of INPP5E^D477N/D477N cilia.

Statistical data are presented as means ± 95% CIs; unpaired t tests (D and L); unpaired t test with Welch’s correction (H) and Mann-Whitney tests (P); n = 3 (control) and n = 2 (mutant) lines for (D), (L), and (T); n = 45 (control) and n = 30 (mutant) cilia from three and two different lines, respectively (H, P, and X); ^∗∗p < 0.01; ^∗∗∗p < 0.001; ^∗∗∗∗p < 0.0001. Scale bar, 2.5 μm. See also Figures S6 and S7.