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. 2023 May 15;133(10):e165019. doi: 10.1172/JCI165019

Figure 6. CERT gain of function causes neurological defects in D. melanogaster.

Figure 6

(A) Schematic of transgenic dCERT flies on the w1118 background. dCERTWT, +WT; dCERTp.S149L, +SL; Chr, chromosome. (B) Quantification of endogenous or exogenous dCERT mRNA levels in flies on the w1118 background (Ctrl) in dCERT-transgenic flies. Data indicate the log2 fold change over Ctrl or +WT (n = 6; data are the mean ± SD). (C) Representative specimens of Ctrl, +WT, and +SL adult flies. Head length (HL), abdomen length (AL), and wing length (WL) were measured. Scale bars: 1 mm. (D) 3D rendering of a micro-CT scan of the heads from Ctrl, +WT, and +SL adults (frontal view). Brain volume is highlighted in pink. Body axes are dorsal (D) and left (L). Scale bars: 100 μm. (E) Z-projections of confocal stacks of whole-mount adult Ctrl, +WT, and +SL adult brains (frontal view) immunostained with anti-nc82. Scale bars: 100 μm. (F) HL/AL ratio of flies reared on DMSO or 10 μM HPA-12 (HPA) (Ctrl, n = 26 or 30, +WT n = 31 or 31, and +SL, n = 25 or 38). (G) Brain volume for Ctrl, +WT, and +SL flies as determined by confocal microscopy (n = 3–4; data are the mean ± SEM). (H) Mass spectrometry profile of sphingolipids in Ctrl, +WT, and +SL adult heads (n = 8). (I) Locomotion of Ctrl, +WT, and +SL flies plotted as total counts per 30 minutes over time (n = 16). (J) Locomotion of flies pretreated with 0 or 10 μM HPA-12 (n = 12). (K) Climbing ability of flies at 2, 9, 16, and 23 days post eclosion (dpe) after vigorous mechanical stress (n = 9). Data shown are the mean ± SEM. *P < 0.05, **P < 0.01, ***P < 0.001, and ****P < 0.0001, by 1-way ANOVA.