FIGURE

Fig. 3

ID

ZDB-FIG-251104-12

Publication

Mero et al., 2025 - HPDL Biallelic Variants in Cerebral Palsy and Childhood-Onset Hereditary Spastic Paraplegia: Human and Zebrafish Insights

Other Figures

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Fig. 3

In vivo studies in zebrafish. (A) Burst activity in zebrafish embryos at 30 hpf (hours post-fertilization) is decreased in hpdl-F0 versus controls (hpdl-F0, n = 70; ctrls, n = 83). (B) Automated analysis of spontaneous motor activity revealed an increase in swim distance and velocity in hpdl-F0 at 120 hpf compared with control siblings (hpdl-F0, n = 106; ctrls, n = 106). (C) Detection of dying cells by acridine orange staining in head and tail of control and hpdl-F0 embryos at 24 hpf. Arrows indicate the higher number of dying cells in hpdl-F0 embryos compared with controls. (D) (left) Representative fluorescence images of reactive oxygen species (ROS) generation in zebrafish larvae at 48 hpf (left). Quantitative analysis of ROS production (right). In all graphs, asterisks denote statistically significant differences: *P ≤ 0.05, **P ≤ 0.01.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB + CRISPR1-hpdl
Knockdown Reagent:	CRISPR1-hpdl
Observed In:	head apoptotic process head reactive oxygen species locomotion post-vent region apoptotic process swimming behavior
Stage Range:	Prim-5 to Day 5

Phenotype Detail

Acknowledgments

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