FIGURE

Fig. 5

ID

ZDB-FIG-130129-21

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. 5

Mannose supplementation rescues mpi morphants. (A) Phenotype rescue of mpi morphants treated with 50 mM mannose at 4 dpf. Liver visualized using Tg(fabp10:dsRed). Scale bar: 100 μm. (B) 77% of mpi morphants treated with 50 mM mannose from fertilization through 4 dpf appear completely normal, whereas no rescue is seen with glucose. *P<0.0001 by Fisher′s exact test as compared with mpi MO + water or mpi MO + glucose supplementation. (C) Liver size is improved in mpi morphants treated with mannose supplementation. *P<0.0001 by Fisher′s exact test. (D) Mpi activity is not significantly changed with mannose supplementation. (E) FACE analysis with quantification showing restoration of G₃M₉ LLO levels in mpi morphants treated with mannose as compared with mpi MO alone. Average of two experiments. (F) LLO and M6P levels are increased in mpi morphants treated with mannose to 105% and 68%, respectively. Average of two experiments. Numbers in parentheses under n values indicate the number of experiments.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	gz15Tg + MO1-mpi
Condition:	chemical treatment by environment: mannose
Knockdown Reagent:	MO1-mpi
Observed In:	liver
Stage:	Day 4

Phenotype Detail

Acknowledgments

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