ZFIN ID: ZDB-FIG-170605-16
Boyd et al., 2017 - Bioenergetic status modulates motor neuron vulnerability and pathogenesis in a zebrafish model of spinal muscular atrophy. PLoS Genetics   13:e1006744 Full text @ PLoS Genet.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Gene:
Fish:
Knockdown Reagent:
Anatomical Term:
Stage: Prim-15
PHENOTYPE:
Fish:
Condition:
Knockdown Reagent:
Observed In:
Stage: Prim-15

Fig. 6

Overexpression or pharmacological activation of pgk1 rescues motor neuron phenotypes in smn morphant zebrafish.

(A) Representative confocal micrographs of primary motor neuron axons exiting the spinal cord in control (top), smn morphant (middle) and smn morphant over-expressing pgk1 (bottom) Tg(hb9:GFP) zebrafish embryos. Note the presence of an axonal outgrowth/branching phenotype associated with smn knockdown (arrow heads) that is reduced in the pgk1 over-expressing animals. Scale bars = 50 μM. (B) Overexpression of Pgk1 in smn morphant zebrafish at 30 hpf led to a significant increase in normal motor axons and significant decrease in severe axonal outgrowth phenotypes compared to single smn MO injected embryos. (C) Representative confocal micrographs of motor neuron axons exiting the spinal cord in control (top), smn morphant (middle) and smn morphant treated with 2.5 μM terazosin (bottom) Tg(hb9:GFP) zebrafish embryos. Note how the presence of the axonal outgrowth/branching phenotype associated with smn knockdown (arrow heads) is reduced in the terazosin-treated animals. (D) Bar chart (mean & s.e.m) showing activation of Pgk1 by treatment with 2.5 μM terazosin in smn morphant zebrafish at 30 hpf led to a significant increase in normal motor axons and significant decrease in severe axonal outgrowth phenotypes compared to untreated smn MO injected embryos. Unpaired two-tailed student t-tests * p<0.05, ** p<0.01 *** p<0.001. n = 20 embryos per group.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
GFP ml2Tg standard conditions Prim-15 primary motor neuron IFL
ml2Tg + MO1-smn1 standard conditions Prim-15 primary motor neuron IFL
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
ml2Tg + MO1-smn1 standard conditions Prim-15 primary motor neuron axon decreased length, abnormal
Prim-15 primary motor neuron axon terminus swollen, abnormal
Prim-15 primary motor neuron axonogenesis disrupted, abnormal
ml2Tg + MO1-smn1 chemical treatment: terazosin Prim-15 primary motor neuron axon length, ameliorated
Prim-15 primary motor neuron axonogenesis process efficacy, ameliorated
Acknowledgments:
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