FIGURE

Fig. 3

ID
ZDB-FIG-170620-22
Publication
Vieira et al., 2017 - Repression of phosphatidylinositol transfer protein α ameliorates the pathology of Duchenne muscular dystrophy
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Fig. 3

Pitpnaa knockdown rescues sapje zebrafish muscle phenotype, improves swim velocity, and increases long-term survival. (A) Birefringence assay (Upper) and immunofluorescent staining for myosin heavy chain (Lower) showing the typical abnormal muscle phenotype of affected homozygous-null sapje fish at 4 dpf. Injection of pitpnaa MO at the one-cell stage prevented manifestation of the muscle phenotype in some homozygous-null fish, termed escaper fish, which showed healthy muscle morphology comparable to normal fish. [Scale bars: 1 mm (Upper), 100 μm (Lower).] (B) Percent of affected sapje fish as determined by birefringence assay at 4 dpf. Mating of heterozygous sapje fish normally yields ∼25% of affected embryos. Cohorts injected with 1 ng of pitpnaa MO at the one-cell stage showed significantly lower percentages of affected fish compared with control MO. Ten replicate experiments were performed, n = 200–300 per replicate. Bars represent means ± SDM. *P < 0.0001 by Student’s t test. (C) Swim velocity of sapje fish tracked on the DanioVision system. Injection with 1 ng of pitpnaa MO increased swim velocity of affected fish during a 15-min tracking period performed at 4 dpf. Bars represent means ± SEM; *P < 0.05 by two-way ANOVA and Bonferroni post hoc test. (D) Long-term survival assay showing increased survival of affected fish injected with 1 ng of pitpnaa MO at the one-cell stage. Affected fish were identified by birefringence assay at 4 dpf and followed until 30 dpf. Data represent means ± SDM. *P < 0.05, **P < 0.002 versus noninjected by Student’s t test.

Expression Data
Antibody:
Fish:
Knockdown Reagent:
Anatomical Term:
Stage: Day 4

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Knockdown Reagent:
Observed In:
Stage: Day 4

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 @ Proc. Natl. Acad. Sci. USA