Flies that model PCH1b amino acid substitutions in Drosophila show a range of morphological and behavioral phenotypes.

(A) Rrp40 mutant flies show a progressive wing position phenotype in adults. Abnormal wing posture phenotypes (wings held-up or -out) are shown for Rrp40 mutant flies compared to Rrp40-WT flies with Days post-eclosion indicated for each representative image. (B) Quantitative analysis of the abnormal wing position phenotype of Rrp40 mutants raised at 25°C. The percentages of adults with abnormal wing position over time (Days post-eclosion) were calculated based on 60 flies per genotype. Neither w¹¹¹⁸ nor Control Rrp40-WT flies showed any abnormal wing position (0% abnormal wing posture, n>100) over the time course analyzed. (C) Locomotor activity was assessed using a negative geotaxis assay as described in Materials and methods. Data are presented as the average percentage of flies of the indicated genotypes that reach the top of a cylinder after 60 seconds across all trials. Groups of 10 age-matched flies [Day (1) and Day (6)] were tested for at least three independent trials per genotype. Values represent the mean ± SEM for n = 3 independent experiments. Asterisks (*) indicate results that are statistically significance at *p-value < 0.05; **p<0.01. Results that show no statistical significance when compared are indicated by NS.