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. 2023 Feb 22;8(4):e166073. doi: 10.1172/jci.insight.166073

Figure 2. Absence of UBE3A leads to stalled striatal neurodevelopment.

Figure 2

(A) Representative voltage responses at indicated postnatal days obtained with 3 current injections: 1) hyperpolarizing (–100 pA), 2) subthreshold depolarizing (+50 pA), and 3) over-threshold depolarizing (+250 pA) currents showing changes of input resistance at hyperpolarized and depolarized domains, and of firing rates, respectively. (B) F-I curves, 1-way RM ANOVA (P15: F1,34 = 0.28, P = 1.00; P21: F1,50 = 2.36, P = 0.0005; P35: F1,34 = 2.70, P = 0.0005; P45: F1,50 = 4.65, P = 0.0005; P130: F1,49 = 7.90, P = 0.0005). (C) Rheobase, factorial ANOVA (F4,231 = 3.42, P = 0.01, see Supplemental Table 1). (D) Capacitance, factorial ANOVA (F4,231 = 1.73, P = 0.14), with age (F4,231 = 14.74, P = 0.0001) and genotype (F1,231 = 23.611, P = 0.0001). (See Supplemental Table 1.) (E and F) Input resistance at depolarized (E) and hyperpolarized (F) domains, factorial ANOVA (in E, F4,231 = 1.64, P = 0.165; and in F, F4,231 = 0.09, P = 0.984), with age (in E, F4,231 = 7.156, P = 0.0001; and in F, F4,231 = 48.188, P = 0.0001) and genotype (in E, F1,231 = 10.363, P = 0.0001; and in F, F1,231 = 23.187, P = 0.0001). (See Supplemental Table 1.) (G) Representative recordings of sEPSCs at indicated postnatal days. (H) sEPSC frequency, factorial ANOVA (F4,205 = 2.87, P = 0.024). Sample size (N = neurons/mice) (P15: WT: N = 13/2; LSL: N = 13/2; P21: WT: N = 37/5; LSL: N = 38/5; P35: WT: N = 17/3; LSL: N = 13/2; P45: WT: N = 21/3; LSL: N = 26/4; P130: WT: N = 27/7; LSL: N = 21/5). Data represent mean ± SEM. *P ≤ 0.05, **P ≤ 0.01, ***P ≤ 0.001, ****P ≤ 0.0001. See also Supplemental Table 1.