Fig. 1

(A) Homozygosity plot of dory^p177 mutants based on whole genome sequencing results. Homozygosity scores close to 1.0 indicate linkage to TL alleles while scores close to 0.0 indicate linkage to WIK alleles. Asterisk indicates the position of the identified splice-donor site mutation in cacna2d3 on chromosome 11. (B) Schematics of the cacna2d3 gene and the amino acid sequences encoded by the wild-type cacna2d3 allele and the dory^p177 mutant allele. In the cacna2d3 gene diagram, the site of the point mutation in dory^p177 mutants is indicated by a red asterisk. (C) Schematic representation of the acoustic startle habituation assay. Larvae were exposed to 10 “sub-threshold” low-intensity acoustic stimuli delivered at 30s interstimulus intervals (ISI) to access startle sensitivity. Next larvae were exposed to 10 high-intensity non-habituating stimuli delivered at 30s ISI to determine baseline startle responsiveness followed by 30 high intensity habituating stimuli at a 3s ISI to access habituation. The blue plot was generated by pooling control animal responses to demonstrate a typical wild-type response at each phase of the assay. (D) Mean acoustic startle responsiveness of wild-type (shown in blue) and dory^p177 mutants (shown in red) to each of the stimuli presented during the habituation assay. (E) Mean acoustic startle habituation percentage is calculated by taking the ratio of the mean frequency of startle responsiveness (startle probability) of each larva to stimuli 41–50 over stimuli 11–20. (F) Mean acoustic startle habituation percentage of wild-type and dory^p177 mutants. Wild-type larvae are shown in blue and homozygous dory^p177 mutants are shown in red. Number of larvae shown below each bar. ****p<0.0001, Mann-Whitney test versus wild-type. Error bars indicate SEM.