Fig 1: UNC5B and DCC silencing partially reverse neurite and growth cone defects in UBQLN24XALS inducible motor neurons (iMNs).(A) UBQLN24XALS iMNs expressing shNT, shUNC5B, or shDCC were subjected to Sholl analysis of neurite length and complexity as described in Figure 7. Data analysis was performed using ordinary one-way ANOVA. Data are shown as mean ± SEM. n>100 iMNs, *p=0.05, **p=0.01, ***p=0.001, ****p=0.0001. (B) Growth cone morphologies in UBQLN24XALS iMNs. UBQLN2WT, UBQLN2P497H, and UBQLN24XALS iMNs. iMNs of the indicated genotypes were stained with a-UBQLN2, a-Tuj1, and phalloidin. Note reduced growth cone elaboration in UBQLN24XALS iMNs. (C) Enhanced growth cone elaboration in UBQLN24XALS iMNs expressing DCC or UNC5B shRNAs. iMNs of the indicated genotype were stained with a-UBQLN2, a-Tuj1, and phalloidin. Arrowheads denote growth cones. (D) Quantification of growth cones in UBQLN2WT, UBQLN2P497H, and UBQLN24XALS, and UBQLN24XALS iMNs expressing the indicated shRNAs. Data analysis was performed using ordinary one-way ANOVA. Data are shown as mean ± SEM. n>100 iMNs, *p=0.05, **p=0.01, ***p=0.001, ****p=0.0001. Figure 9—source data 1.Data sets corresponding to Figure 9A, D.