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. 2018 Feb 1;102(2):278–295. doi: 10.1016/j.ajhg.2018.01.006

Figure 4.

Figure 4

Reduced Expression of OTUD7A Contributes to Spine and Dendrite Defects in Df(h15q13)/+ Mice

(A) Representative images of Venus-expressing dendritic spines from P22 WT or Df(h15q13)/+ neurons co-expressing Venus and pcDNA control or FLAG-OTUD7A WT (scale bar = 5 μm).

(B) Expression of OTUD7A WT rescues dendrite spine density defects in Df(h15q13)/+ cortical neurons. WT + pcDNA control, n = 30 dendrite segments; [Df(h15q13)/+] + pcDNA, n = 30 dendrite segments; [Df(h15q13)/+] + OTUD7A WT, n = 30 dendrite segments; 3 cultures, one-way ANOVA followed by Tukey’s post hoc test, ∗p < 0.05, ∗∗∗p < 0.001, F(2, 87) = 8.049.

(C and D) Df(h15q13)/+ neurons show a decrease in mushroom spines and an increase of stubby spines compared to WT neurons. Expression of OTUD7A WT in Df(h15q13)/+ neurons decreases the proportion of stubby spines compared to Df(h15q13)/+ neurons. WT + pcDNA control, n = 30 dendrite segments; [Df(h15q13)/+] + pcDNA control, n = 30 dendrite segments; [Df(h15q13)/+] + OTUD7A WT, n = 30 dendrite segments; 3 cultures, one-way ANOVA followed by Tukey’s post hoc test, ∗p < 0.05, ∗∗∗p < 0.001, F(2, 87) = 4.249, F(2, 87) = 8.636.

(E) Df(h15q13)/+ neurons showed a decrease in spine length compared to WT neurons, and expression of OTUD7A WT in Df(h15q13)/+ neurons increased spine length. WT + pcDNA control, n = 586 spines; [Df(h15q13)/+] + pcDNA control, n = 475 spines; [Df(h15q13)/+] + OTUD7A WT, n = 664 spines; 3 cultures, one-way ANOVA followed by Tukey’s post hoc test, ∗p < 0.05, ∗∗∗p < 0.001, F(2, 1722) = 3.956.

(F) Representative images of littermate P22 WT and Df(h15q13)/+ neurons expressing Venus and pcDNA control or OTUD7A WT (scale bar = 50 μm).

(G) Expression of OTUD7A WT rescues dendrite growth defects (number and total number of intersections) in Df(h15q13)/+ cortical neurons. WT + pcDNA control, n = 28 neurons, [Df(h15q13)/+] + pcDNA control, n = 24; [Df(h15q13)/+] + OTUD7A WT, n = 28 neurons; 3 cultures, one-way ANOVA followed by Tukey’s post hoc test, ∗p < 0.05, ∗∗∗∗p < 0.0001, F(2, 61) = 13.53. Error bars represent SEM.