Peripheral neuropathy phenotypes induced by hGlyRS-S211F and hGlyRS-H418R are not attributable to a dominant negative mechanism. (A), Negative geotaxis climbing speed of 7-day-old female flies expressing hGlyRS-S211F or hGlyRS-H418R transgenes in motor neurons (OK371-GAL4), in the presence or absence of dGlyRS co-overexpression. n = 10–35 groups of 10 flies per genotype; ***P< 0.0001 by Kruskal–Wallis test with Dunn’s multiple comparisons test. (B), Synapse length on distal muscle 8 of larvae expressing hGlyRS-S211F or hGlyRS-H418R transgenes in motor neurons (OK371-GAL4), in the presence or absence of dGlyRS co-overexpression. n = 10–12 animals per genotype; ***P< 0.0001 by one-way ANOVA with Sidak’s multiple comparisons test. (C), Quantification of dendritic coverage (% of driver-only control) of class IV multidendritic sensory neurons in the larval body wall upon selective expression of hGlyRS-S211F or hGlyRS-H418R transgenes in these neurons (ppk-GAL4), in the presence or absence of dGlyRS co-overexpression. n = 11–12 animals per genotype; ***P< 0.005 by one-way ANOVA with Sidak's multiple comparisons test. (Dand E), Quantification (D) and representative images (E) of relative translation rate (% of driver-only control) as determined by THRONCAT in motor neurons (OK371-GAL4) of larvae expressing hGlyRS-S211F or hGlyRS-H418R, in the presence or absence of dGlyRS co-overexpression. Motor neurons targeted by OK371-GAL4 are delineated by a dashed line and nuclei of motor neurons are labeled by a white dot (E). n = 8–13 animals per genotype; ***P< 0.0001 by one-way ANOVA with Sidak’s multiple comparisons test. Scale bar: 10 μm.