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Fig. 1 | Acta Neuropathologica Communications

Fig. 1

From: Liver X receptor-agonist treatment rescues degeneration in a Drosophila model of hereditary spastic paraplegia

Fig. 1

Loss of Arl6IP1 results in a progressive locomotor deficit in vivo. a Partial sequence of Drosophila Alr6IP1 showing the location of the sgRNA and PAM site used (top panel). (Lower panels) DNA sequencing results of CRISPR control and Arl6IP1 knockout (KO) lines generated in this study aligned using Geneious software. b Graph represents mean ± standard error from the mean (SEM) real-time PCR analysis of Arl6IP1 expression in CRISPR control and Arl6IP1 KO Drosophila. Statistical analysis consists of one-way ANOVA and subsequent Dunnett’s post-hoc tests. n = 3. c Representative confocal images of the posterior NMJ stained with the post-synaptic density marker DLG (magenta). d Graph represents mean ± SEM number of 1b, 1 s, and total synaptic boutons in control and Arl6IP1 KO. n = 19–22 animals. Statistics consist of two-way ANOVA with Bonferroni’s post-hoc tests. e Arl6IP1 knockout flies exhibit locomotor deficits that progress over time. Graph represents the mean ± SEM percentage of flies that reached the top of the vial within 15 s. Statistical analysis consists of two-way ANOVA and Dunnett’s multiple comparisons tests. n = 13—16 independent experiments per genotype. f Survival assay reveals no effect of loss of Arl6IP1 on lifespan compared to controls. n = 115–126 flies per genotype. p-value = 0.6967 (ns) determined by Log-rank (Mantel-Cox) test

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