Figure 2.

Figure 2. The I209N mutation in Nsfa selectively affects hearing and vestibular function without causing paralysis

(A) Still frames from high-speed videos of larvae. nsfa^st53 homozygotes were paralyzed and unresponsive to touch, whereas nsfa^I209N homozygotic and nsfa^I209N/st53 larvae displayed robust startle reflexes.

(B) (B and C) Acoustic evoked behavioral responses of larvae exposed to a 600-Hz stimulus at the intensities indicated normalized to homozygous WT siblings. n indicates number of fish tested.

(D–K) Vestibulospinal reflexes are reduced in nsfa^I209N homozygotic and nsfa^I209N/st53 larvae. (E–G) Raw traces (blue) of the tail movements of representative WT and mutant larvae. The rolling median (orange trace) is a movement artifact. (H–K) The maximum tail angle (H and I) and the normalized integral (J and K) of sibling cohorts were quantified using ZebraZoom. n indicates number of fish tested.

Mean ± SEM and two-way ANOVA with Benjamini-Hochberg correction for each dataset were performed. *p < 0.05, **p < 0.01, ***p < 0.001.