FIGURE

Fig. 2

ID

ZDB-FIG-201113-23

Publication

Cook et al., 2020 - Visualisation of cholesterol and ganglioside GM1 in zebrafish models of Niemann-Pick type C disease and Smith-Lemli-Opitz syndrome using light sheet microscopy

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Fig. 2

Identification of SLOS phenotypes in Dhcr7 inhibitor and dhcr7 morpholino zebrafish larvae models. SLOS phenotypes were induced in the zebrafish larvae using either Dhcr7 inhibitors AY9944 (75 µM) or trazodone (traz, 75 µM), or dhcr7 morpholino (dhcr7 MO) injected at the 1–2 cell stage with DMSO and Sham controls as necessary; a Morphology of 72 hpf zebrafish by light microscopy. Arrowheads indicate heart defects. Quantitative analysis of 72 hpf zebrafish by light microscopy; b eye area. Quantitative behavioural analysis of 72 hpf inhibitor treated zebrafish; c heart rate over 1 min and d escape response time. Quantitative behavioural analysis of 72 hpf dhcr7 morphant zebrafish; e heart rate over one minute and f escape response time. All analysis by Kruskal–Wallis test with Dunn’s post-hoc correction (b, c, d) or one-way ANOVA with Tukey post-hoc correction (e, f). *p < 0.05, **p < 0.01, ****p < 0.0001. Scale bars = 280 µm. N = 3, with a minimum of 3–10 fish per N

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB/TL AB/TL + MO1-dhcr7
Conditions:	chemical treatment: N-[(2-chlorophenyl)methyl]-1-[4-[[(2-chlorophenyl)methylamino]methyl]cyclohexyl]methanamine chemical treatment: trazodone
Knockdown Reagent:	MO1-dhcr7
Observed In:	eye heart contraction pericardium psychomotor behavior
Stage:	Protruding-mouth

Phenotype Detail

Acknowledgments

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