FIGURE

Fig. 3

ID

ZDB-FIG-130129-19

Publication

Chu et al., 2013 - A zebrafish model of congenital disorders of glycosylation with phosphomannose isomerase deficiency reveals an early opportunity for corrective mannose supplementation

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Fig. 3

mpi morphants develop multi-systemic abnormalities. (A) Injection of 6.7 ng of mpi MO results in morphants that are characterized by a small head, microphthalmia, pericardial edema, jaw defects and reduced liver size as visualized in live fish expressing dsRed in hepatocytes [Tg(fabp10:dsRed)] from 30 to 100 hpf. Scale bar: 50 μm. (B) Embryo clutches injected with mpi MO have an average of 15% phenotypically normal embryos as compared with 93% of control embryos. Range from 0–35% normal. P<0.0001 by Fisher′s exact test. Numbers in parentheses under n values indicate the number of experiments. (C) Injection of 6.7 ng of mpi MO results in abnormal liver development. Liver size correlated with severity of phenotype. (D) ‘Phenotype score’ equation. (E) Linear regression analysis shows that phenotype score is inversely correlated with residual Mpi enzyme activity.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB + MO1-mpi gz15Tg + MO1-mpi
Knockdown Reagent:	MO1-mpi
Observed In:	eye forebrain liver mandibular arch skeleton pericardium post-vent region
Stage Range:	Long-pec to Day 4

Phenotype Detail

Acknowledgments

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