ZFIN ID: ZDB-FIG-170220-17
Linsley et al., 2017 - Congenital myopathy results from misregulation of a muscle Ca2+ channel by mutant Stac3. Proceedings of the National Academy of Sciences of the United States of America   114(2):E228-E236 Full text @ Proc. Natl. Acad. Sci. USA
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Knockdown Reagent:
Observed In:
Stage: Day 4

Fig. 2

DHPR tetrads are reduced and incomplete in stac3 mutants. (AE) Freeze-fracture electron micrographs of 4-d postfertilization larvae showing DHPR particles in triadic clusters of WT (A), stac3−/− expressing stac3WT-EGFP (WT rescue) (B), stac3−/− expressing stac3NAM-mKate2 (NAM rescue) (C), stac3−/− (D), and stac3−/− injected with an antisense morpholino oligonucleotide against stac3 (stac3−/− + MO) (E). (A′E′) Same as AE, with yellow dots and purple shading added for clarity to denote, respectively, segments of T tubules with tetrad sites of DHPRs and segments of T tubules with no tetrad sites in muscle fibers of WT (A′), WT rescue (B′), NAM rescue (C′), stac3−/− (D′), and stac3−/− + MO (E′). (F) Illustration showing stereotypical DHPR particles in tetrad sites (labeled with yellow dots) along a T tubule and gaps with no tetrad sites (purple) as seen above. (G) Histogram showing that the particles per T-tubule length are decreased in NAM rescue, stac3−/−, and stac3−/− + MO muscles compared with WT and WT rescue. (ANOVA Tukey's; ***P < 0.001, **P < 0.01.) (H) Histogram showing that full tetrads per tetrad site are decreased in NAM rescue, stac3−/−, and stac3−/− + MO muscles compared with WT and WT rescue. ns, not significant. SEMs are indicated. (ANOVA Tukey's; ***P < 0.001, *P < 0.05.)

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
mik34wtTg ; stac3mi34/mi34 control Day 4 muscle cell L-type voltage-gated calcium channel complex amount, ameliorated
stac3mi34/mi34 control Day 4 muscle cell L-type voltage-gated calcium channel complex decreased amount, abnormal
stac3mi34/mi34 + MO2-stac3 control Day 4 muscle cell L-type voltage-gated calcium channel complex decreased amount, abnormal
stac3mi34/mi34 ; mik34namTg control Day 4 muscle cell L-type voltage-gated calcium channel complex decreased amount, abnormal
Acknowledgments:
ZFIN wishes to thank the journal Proceedings of the National Academy of Sciences of the United States of America for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Proc. Natl. Acad. Sci. USA