Fig. 8: Ankle and knee joints show hyperflexion in SOD1G93A mice and after silencing of En1 positive neurons.
a Photograph showing the lateral view of a mouse analyzed with DeepLabCut tracking software. Dots mark iliac crest, hip, knee, ankle, foot and toe. Stick figures depicting the relative position of the analyzed joints in wild-type (b) and in SOD1G93A mice after Onset of locomotor phenotype (c) during a complete step cycle, stance phase in gray. Stick figures of En1cre;HoxB8FlipO; RC::Di mice before (d) and after (e) CNO treatment show similar changes in hyperflexion of the ankle and knee joints as in the SOD1G93A mice after onset. Quantification of the changes in angles of hip in wild-type versus SOD1G93A conditions (f–g, j) show no differences (two-tailed t test, P = 0.2059, t = 1.344, df = 11), while knee (l–m, p) (two-tailed t test, P = 0.0196, t = 2.729, df = 11), ankle (r–s, v) (two-tailed t test, P = 0.0483, t = 2.221, df = 11) and foot (x–y; ab) (two-tailed t test, P = 0.0052, t = 3.469, df = 11) are significantly different between the two conditions (wt N = 8 mice, SOD1G93A N = 5 mice). Quantifications of the joint angles after CNO administration mimics the SOD1G93A mice after onset. While no changes were observed in the hip angle (h–i, k) (two-tailed t test, P = 0.7901, t = 0.2722, df = 12) angles of knee (n–o, q) (two-tailed t test, P = 0.0212, t = 2.650, df = 12), ankle (t–u; w) (two-tailed t test, P = 0.0140, t = 2.873, df = 12) and foot (z-aa; ac) (two-tailed t test, P = 0.0071, t = 3.239, df = 12) were significantly different before and after CNO treatment (En1cre;HoxB8FlipO;RC::Di N = 7 independent mice). In all graphs, data are presented as mean values ± SEM. Error bands in (f–i), (l–o), (r–u) and (s-aa) represent SD. Source data are provided as a Source Data file.
