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.

Other Figures

All Figure Page

Back to All Figure Page

Fig. 8

2HPβCD 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 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 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 μm. (D) Lyso R relative intensity was significantly reduced in npc1^m/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 npc1^m/m larvae treated with 2.5 mM 2HPβCD. Scale bar: 200 μm.

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

This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Dis. Model. Mech.