FIGURE

Fig. 2

ID

ZDB-FIG-230829-13

Publication

Weinschutz Mendes et al., 2023 - High-throughput functional analysis of autism genes in zebrafish identifies convergence in dopaminergic and neuroimmune pathways

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Fig. 2

Behavioral phenotypes in zebrafish ASD gene mutants

(A and B) Experimental setup and behavioral assays. Visual-startle: Larvae are exposed to five 1-s flashes of lights-off or -on stimuli at 29-s intervals at 5 dpf. Sleep-wake: Larvae are exposed to a 14 h:10 h white light:dark schedule at 5?7 dpf. Visual-startle and sleep-wake parameters are shown (see STAR Methods).

(C) Hierarchical clustering of mutant behavioral fingerprints. Each rectangle represents the signed -log₁₀-transformed p values from linear mixed models (LMM) comparing mutant and background-matched wild-type fish (red, increased in mutant; blue, decreased in mutant). Behavioral parameters from startle-off (light blue), startle-on (orange), and sleep-wake (purple) are shown. All mutant lines are shown except katnal2, which lacks consistently significant behavioral features (Figure S2A). Three mutant lines andtransheterozygotes (scn1lab^?44/?5) are shown for scn1lab. Behavioral features shown in individual graphs (D?G) are outlined in cyan. Mean and SD values for all groups and beta and p values for all behavioral parameters are shown in Table S2.

(D?G) Significant behavioral phenotypes in ASD gene mutants: increased responses to lights-on stimuli, daytime hypoactivity, and night-time hyperactivity in scn1lab^?44/?44 (D); decreased responses to lights-off stimuli and daytime hypoactivity in dyrk1aa^?77/?77dyrk1ab^?8/?8 (E); decreased daytime sleep bout length in chd8^?5/?5 (F); and daytime hyperactivity in tbr1a^?64/?64tbr1b^?10/?10 (G). Sleep-wake time series graphs in (D?G) show a rolling average of activity data points every 50 min. ^????p < 0.0001, ^???p < 0.001, ^??p < 0.01 (one-way ANOVA).

(H) Correlation analysis across all 24 behavioral parameters identifies three sub-groups with related phenotypes: (i) scn1lab^?44/?44, chd8^?5/?5, cntnap2a^?121/?121cntnap2b^31ins/31ins; (ii) pogza^?23/?23pogzb^?20/?20, dyrk1aa^?77/?77dyrk1ab^?8/?8, cul3a^?7/+cul3b^?32/?32; and (iii) tbr1a^?64/?64tbr1b^?10/?10, kdm5ba^?17/?17kdm5bb^?14/?14, grin2ba^?25/?25grin2bb^?64/?64.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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