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ZFIN ID: ZDB-FIG-130129-19
Chu et al., 2013 - A zebrafish model of congenital disorders of glycosylation with phosphomannose isomerase deficiency reveals an early opportunity for corrective mannose supplementation. Disease models & mechanisms   6(1):95-105 Full text @ Dis. Model. Mech.
Knockdown Reagent:
Observed In:
Stage Range: Long-pec to Day 4

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.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
AB + MO1-mpi standard conditions Long-pec - Pec-fin eye decreased size, abnormal
Long-pec - Pec-fin forebrain decreased size, abnormal
Long-pec - Pec-fin mandibular arch skeleton malformed, abnormal
Long-pec - Pec-fin pericardium edematous, abnormal
Long-pec - Pec-fin post-vent region curved, abnormal
gz15Tg + MO1-mpi standard conditions Day 4 liver decreased size, abnormal
ZFIN wishes to thank the journal Disease models & mechanisms for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Dis. Model. Mech.