Fig. 3.

Anks1b heterozygous mice recapitulate phenotypes in ANKS1B haploinsufficiency syndrome. a In the Behavioral Spectrometer, Nestin-Het mice covered more track in 9 min and had more episodes of running than Nestin-WT controls. Reduction in grooming behavior was not statistically significant. Nestin-Het mice visit the center square of an open field more often than controls. b Nestin-Het mice showed a robust reduction in avoidance behaviors, covering more track in the open arms as a percentage of total track in open and closed arms of an elevated plus maze. c In the three-chamber test, Nestin-Het mice show significantly reduced and borderline preference for a conspecific mouse over an inanimate object. d Peak magnitude of the acoustic startle reflex is robustly increased in Nestin-Het mice. Sensorimotor gating measured by the percentage of prepulse inhibition (prepulse stimulus 40 ms before startle stimulus) is significantly reduced. e In a test of fine-motor dexterity, Nestin-Het mice require more time to remove adhesive from the forepaw. Nestin-Het mice do not exhibit significant deficits in gross motor coordination as measured by slips on a balance beam. f Nestin-Het mice do not show learning deficits in the object placement test, a hippocampus-dependent memory assay. Both groups averaged above a passing score (>50% preference for new location) using a 40-min retention interval. Raising the task difficulty by increasing the retention interval to 90 min did not result in a difference: mean values indicate that mice of both genotypes failed the test at 90 min (<50% preference). Reference line indicates 50% preference. Box plots show the mean and 95% confidence intervals (black diamond), median (black line), 25th–75th quantile (gray or red bar), and range (black whiskers). If two-way ANOVA showed significant main effect of genotype, post hoc two-sided Student’s t-test was performed, *p < 0.05, **p < 0.01, ***p < 0.001, ns = p > 0.05. g Numbers and sex of mice tested with the measures for each behavioral assay are shown as the mean and SEM. No sex-dependent effects of genotype were observed in any behavioral test by two-way ANOVA (Supplementary Data 5)