FIGURE

Fig. 1

ID

ZDB-FIG-210426-29

Publication

Cinzia et al., 2021 - Therapeutic efficacy of 3,4-Diaminopyridine phosphate on neuromuscular junction in Pompe disease

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Fig. 1

3,4-DAPP administration increased density and appropriate spatial positioning of AChRs. (A) Representative image of AChRs (α-BTX) in 4 somites of zebrafish embryos at 48 hpf collected during 3 distinct experiments. STD-Ctrl embryos: n = 32, untreated I9E10gaa-MO: n = 30, and I9E10gaa-MO treated with 3,4-DAPP 50 µM: n = 31, Scale bar = 25 µm. (B) Quantification of α-BTX-positive spots in the three experimental groups. (C) Representative image of single myofibers obtained after dissociation from 3 dpf embryos. Scale bar = 30 µm. (D,E) Quantification of AChR clusters in dissociated myofibers (D), and of myofiber average length (E) in the three experimental groups. Note that myofiber average lengths are comparable. Error bars in (B), (D), (E) are SEMs.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB + MO2-gaa
Condition:	chemical treatment by environment: amifampridine
Knockdown Reagent:	MO2-gaa
Observed In:	skeletal muscle cell somite skeletal muscle acetylcholine-gated channel clustering
Stage:	Long-pec

Phenotype Detail

Acknowledgments

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