Fig. 8
- ID
- ZDB-FIG-180827-84
- Publication
- Tseng et al., 2018 - Modeling Niemann-Pick disease type C1 in zebrafish: a robust platform for in vivo screening of candidate therapeutic compounds.
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2HPβCD treatment reduces LysoTracker Red and filipin staining in npc1m/m neuromasts. (A) Control and mutant larvae can be differentiated at 3 dpf by intense LysoTracker Red (Lyso R) staining of the olfactory placode (arrowheads) at 3 dpf. Scale bars: 100 μm. (B) Mutant larvae selected by Lyso-R-positive olfactory placode staining were treated for 3 days with vehicle (ddH2O) or 2.5 mM 2HPβCD starting at 3 dpf. Neuromast Lyso R staining was markedly decreased in 2HPβCD-treated npc1m/m larvae. Scale bars: 1 mm. (C) Higher magnification view of Lyso R staining, corresponding to the boxed area in B. Scale bar: 100 μm. (D) Lyso R relative intensity was significantly reduced in npc1m/m larvae treated with 2.5 mM 2HPβCD at 6 dpf. ****P<0.0001; ns, not significant. Significance was calculated by two-tailed t-test. (E) Filipin staining confirmed decreased unesterified cholesterol accumulation in neuromast (arrowheads) of npc1m/m larvae treated with 2.5 mM 2HPβCD. Scale bar: 200 μm. |
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Stage Range: | Protruding-mouth to Day 6 |