FIGURE 3

FIGURE 3

lhfpl5a is required for auditory and vestibular hair cell function. (A) Boxplot showing the first peak amplitude (μV) of microphonic recordings from the inner ear of 3 dpf wild type, lhfpl5a^tm290d, and lhfpl5b^vo35 larvae. The boxes cover the inter-quartile range (IQR), and the whiskers represent the minimum and maximum datapoints within 1.5 times the IQR. Values from individual larvae are indicated by the black dots and outliers indicated by a diamond. Asterisks indicate p < 0.01 (^∗∗) by Welch’s t-test. ns, not significant. (B) Boxplot of the acoustic startle response in 6 dpf wild type and lhfpl5a^tm290d mutants, with or without the GFP-lhfpl5a vo23Tg transgene. Values from individual larvae are indicated by the black dots and outliers indicated by a diamond. Asterisks indicate p < 0.001 (^∗∗∗) by Welch’s t-test. ns, not significant. (C–F’) FM 4–64 dye labeling assay for MET channel activity of inner ear hair cells from 6 dpf larvae [C – Wild type (+/+ or±), D – lhfpl5a^tm290d mutants, E – Wild type GFP-lhfpl5a vo23Tg, F – GFP-lhfpl5a vo23Tg; lhfpl5a^tm290]. (E’,F’) Are images of GFP-Lhfpl5a protein in the bundle of the hair cells shown above in (E,F). n = 2, 3, 12, and 4 for the genotypes in (C–F), respectively. Scale bars = 5 μm and apply to all images.