Perceptive-motor slowness and hyperactivity disorders are characteristic of people with DS (Ekstein et al., 2011; Mason et al., 2015). In animal models, this phenotype is reflected in altered parameters of locomotor activity. To assess the effects of Kcnj6 dose on spontaneous locomotion, behavior in the ‘activity box’ was examined. Locomotor activity was significantly increased in Ts65Dn:Kcnj6+++ vs. 2N:Kcnj6++ mice. This was registered as a significant increase in ambulatory distance (F1,16 = 4.78; p = 0.0001) (Fig. 3, A, red vs. blue bar) and ambulatory time (F1,17 = 4.83; p = 0.0001) (Fig. 3, B). In addition, resting time was reduced (F1,23 = 3.59; p = 0.0001), but velocity did not differ (F1,15 = 1.13; p = 0.14) in Ts65Dn:Kcnj6+++ vs. 2N:Kcnj6++ mice. Decrease in Kcnj6 dose had no effect on locomotor activity in Ts65Dn mice. Indeed, neither the ambulatory distance (F1,24 = 0.14; p = 0.44) nor the ambulatory time (F1,24 = 0.015; p = 0.49) were significantly different comparing Ts65Dn:Kcnj6+++ and Ts65Dn:Kcnj6++− mice (Fig. 3, A, B, red vs. green bar). Likewise, there were no differences in resting time (F1,24 = 0.29; p = 0.39) or velocity (F1,24 = 0.45; p = 0.33).
