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. 2023 Jan 9;32(10):1647–1659. doi: 10.1093/hmg/ddad004

Figure 2.

Figure 2

Administration of L7-Slc9a6-GFP AAV at 5 weeks of age reduces ataxic motor phenotype at later time points. (A) Center of mass traces from representative movements made at 18 weeks of age. Treated animals are grouped on the basis of treatment efficacy. Color scale refers to quantitative severity of ataxia. Blue refers to WT animals, red refers to untreated shakers (shaker + L7-GFP AAV). Percentage of severity from WT to untreated is represented by the shade of purple. (B)–(D) Straightness of gait is computed as a measure of motor coordination. Shown is a ratio of total distance traveled versus displacement, averaged across all rapid movements made in 30 min at each time point. Ratios are shown at (B) 8 weeks, shortly after the typical onset of PC loss, (C) 18 weeks, shortly after the typical completion of PC loss and (D) 25 weeks, at which point the motor symptom progression is typically complete. L7-GFP AAV had no impact on gait, but L7-Slc9a6-GFP AAV reduced motor dysfunction. (E) Straightness of gait ratios are shown for all rapid movements recorded in all rats at 25 weeks. Untreated shakers primarily make uncoordinated movements, whereas treated shakers with a strong effect primarily make coordinated movements.