FIGURE

Figure 2

ID
ZDB-FIG-210617-83
Publication
Douek et al., 2021 - An Engineered sgsh Mutant Zebrafish Recapitulates Molecular and Behavioural Pathobiology of Sanfilippo Syndrome A/MPS IIIA
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Figure 2

sgshΔex5−6 zebrafish exhibit progressive behavioural abnormalities. Behavioural tracking of 5 dpf wild type and sgshΔex5−6 homozygous larvae, examining (A) cumulative distance travelled, (B) wall preference ratio (Tw), and (C) zone intercepts to and from the central arena reflecting exploration behaviour. In (A–C) each point represents an individual larva. (D) Representative movement traces of single larval movement in the imaging arena throughout the recording period for both wild type and sgshΔex5−6 genotypes. Central blue circle indicates region for zone intercept calculations. Scale bar 15 mm. (E) Cumulative distance travelled by adult (6-month-old) wild type and sgshΔex5−6 zebrafish. (F) Average movement velocity between adult wild type and sgshΔex5−6 zebrafish. (G) Cumulative time spent in exploratory zone between adult wild type and sgshΔex5−6 zebrafish. (H) Frequency of interception with exploratory zone boundaries between adult wild type and sgshΔex5−6 zebrafish. Data in (A–C) are presented as mean ± SEM and tested by Mann-Whitney U test, and in (E–H) by unpaired t-test. In all samples **** p < 0.0001 and n.s. p > 0.05; in G ** p = 0.0029 and in H * p = 0.0146. (I) Representative movement traces of single adult movement in the imaging arena throughout the recording period for both wild type and sgshΔex5−6 genotypes. Inner blue rectangle indicates region for exploratory zone calculations. Scale bars 1 cm.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage Range: Day 5 to Adult

Phenotype Detail
Acknowledgments
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