Impaired locomotor activity and muscular defects in OcrlY/− mice. (A–C) Open-field large-arena test was performed during 10 min of two subsequent days in a circular arena with a diameter of 150 cm on a cohort of 16 OcrlY/+and 16 OcrlY/− age-matched male mice. (A) Illustrative pictures representing the trajectories traveled by Ocrl mice during the open field large arena test. (B) Dot plot representing the distance covered by Ocrl mice during the open-field large-arena test. The distance traveled by each mouse is normalized to 1 min observation time and plotted as function of time for each bin (bin = 5 min; split ANOVA effects followed by post-hoc tests between genotype for each bin: Bin1, *P < 0.1; Bin2, *P < 0.05; Bin3, *P < 0.05). (C) Dotted bar graph representing the frequency of resting, lingering and walking episodes exhibited by Ocrl mice during the open field large arena test (split ANOVA effects followed by post-hoc tests between genotype for each state: rest, *P < 0.05; linger, *P < 0.05; walk, *P < 0.05). (D) IntelliCage experiments were performed on a cohort of age matched male Ocrl mice (13 OcrlY/+and 13 OcrlY/−). Dot plot representing the median licking frequency during drinking restriction protocol. Base indicates the licking frequency during the last 24 h of nose-poke adaptation phase (split ANOVA effects followed by post-hoc tests between genotype for each time point: day 1, **P < 0.01; day 2 to last day, *P < 0.05; last day, **P < 0.001). (E) Lean mass/body weight ratio determined by whole body composition analysis in Ocrl mice (n = 6 mice per group). (F) Muscle mass, relative to body weight, of selected hind limb muscles obtained by bilateral dissection of 5 pairs of Ocrl mice (n = 10 values for symmetrical leg muscles). (G) Plasma CK activity in the corresponding five pairs of Ocrl mice (as in F). Plotted data represent mean ± SEM. In (B–D), one-way ANOVA between subject factor genotype: *P < 0.05, **P < 0.01 (on the top of each graph); in (E–G), Mann Withney test, **P < 0.01, ***P < 0.001 relative to OcrlY/+ mice.