FIGURE

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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Fig. 8

2HP?CD treatment reduces LysoTracker Red and filipin staining in npc1^m/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??m. (B) Mutant larvae selected by Lyso-R-positive olfactory placode staining were treated for 3?days with vehicle (ddH₂O) or 2.5?mM 2HP?CD starting at 3?dpf. Neuromast Lyso R staining was markedly decreased in 2HP?CD-treated npc1^m/m larvae. Scale bars: 1?mm. (C) Higher magnification view of Lyso R staining, corresponding to the boxed area in B. Scale bar: 100??m. (D) Lyso R relative intensity was significantly reduced in npc1^m/m larvae treated with 2.5?mM 2HP?CD 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 npc1^m/m larvae treated with 2.5?mM 2HP?CD. Scale bar: 200??m.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	npc1^hg37/hg37
Condition:	chemical treatment by environment: beta-cyclodextrin
Observed In:	neuromast cholesterol neuromast lysosome peripheral olfactory organ lysosome
Stage Range:	Protruding-mouth to Day 6

Phenotype Detail

Acknowledgments

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