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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. 1

Identification of NPC phenotypes in a Npc1 inhibitor and npc1 morpholino zebrafish larvae model. NPC phenotypes were induced in the zebrafish larvae using either the Npc1 inhibitor, U18666A (2 µg/ml), or npc1 morpholino (npc1 MO) injected at the 1–2 cell stage with DMSO and Sham controls as necessary; a Morphology of 48 h post fertilisation (hpf) zebrafish by light microscopy. Quantitative behavioural analysis of 24 hpf zebrafish measuring number of spontaneous coils over 3 min in 2 µg/ml U18666A treated (b) and npc1 morphant (c) zebrafish, *p < 0.05, **p < 0.01, ****p < 0.0001, Kruskal–Wallis test with Dunn’s post-hoc correction. Representative images of LysoTracker Green staining of 96 hpf control and U18666A treated zebrafish larvae eyes with and without 500 µM miglustat co-treatment (d). Scale bars = 280 µm. N = 3–5, with a minimum of 10 fish per N

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Acknowledgments

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