CSMD3-deficient mice exhibit anxiety-like behaviors and motor impairments. A–E, Assessment of anxiety-like behaviors. A, B, OFT. A, Representative automated tracking of a single mouse in the OFT (5 min duration) across the wild-type, CSMD3^−/−, and CSMD3^+/− mice. B, Bar graph shows time spent in the center. F_(2,12) = 0.55, p = 0.5666 and p = 0.8648, CSMD3^−/− versus WT and CSMD3^+/− versus WT, respectively. C–E, EPM test. C, Representative automated tracking of a single mouse in the EPM (5 min duration) across the wild-type, CSMD3^−/−, and CSMD3^+/− mice. D, Bar graph shows time spent in open arms. F_(2,12) = 7.11, p = 0.0073 and p = 0.0628, CSMD3^−/− versus WT and CSMD3^+/− versus WT, respectively. E, Bar graph shows the number of open arms entries. F_(2,12) = 9.00, p = 0.0038 CSMD3^−/− versus WT; p = 0.0186 CSMD3^+/− versus WT. F–I, Assessment of adult motor function. F, Bar graph shows total distance traveled in OFT; CSMD3^−/− versus WT: F_(2,12) = 11.54, p = 0.0063; CSMD3^−/− versus CSMD3^+/−, p = 0.0022; G, Bar graph shows total distance traveled in EPM; CSMD3^−/− versus WT, F_(2,12) = 9.89, p = 0.0023; CSMD3^+/− versus WT, p = 0.0256. H, I, Rotarod test; line graphs show the latency (H) and speed (I) at which the mice fall from the rod: F_(4,24) = 1.90 (H); F_(4,24) = 2.54 (I). J–N, Assessment of the motor function of pups: ambulation score (J); angle to fall (K); latency to fall (L); righting time (M); posture score (N). F_(2,24) = 15.56, p < 0.0001 and p = 0.0144 (J); F_(2,24) = 35.87, p < 0.0001 and p = 0.0012 (K); F_(2,24) = 57.77, p < 0.0001 and p < 0.0001 (L); F_(2,24) = 26.86, p < 0.0001 and p = 0.9990 (M); F_(2,24) = 0.82, p = 0.4307 and p = 0.6822 (N), CSMD3^−/− versus WT and CSMD3^+/− versus WT, respectively. Data are presented as the mean ± SEM; n = 4–6 mice/group (A–I); n = 9 mice/group (J–N); *p < 0.05, **p < 0.01, and ***p < 0.001, one-way ANOVA with Tukey's post hoc test (B, D–G, J–N); two-way ANOVA with Tukey's post hoc test (H, I).