lhfpl5b is required for lateral line hair cell function. (A–C) Representative images of 5 dpf zebrafish larvae (A – Wild type; B – lhfpl5atm290d; C – lhfpl5bvo35) labeled with the MET channel-permeant dye FM 1–43. (D–G) Representative images of individual neuromasts from 2 and 5 dpf wild type and lhfpl5bvo35 larvae labeled with FM 1–43. Dashed lines outline the cluster of hair cells in each neuromast. (H) Quantification of normalized FM 1–43 fluorescence intensity per hair cell of 2 and 5 dpf neuromasts (n = 10 WT, 14 lhfpl5bvo35 NMs at 2 dpf; n = 6 WT, 13 lhfpl5bvo35 NMs each genotype at 5 dpf). The box plots cover the inter-quartile range (IQR), and the whiskers represent the minimum and maximum datapoints within 1.5 times the IQR. Asterisks indicate p < 0.001 (∗∗∗) by Welch’s t-test. (I,J) Rescue of FM dye labeling in lhfpl5bvo35 mutants (n = 7) by the GFP-lhfpl5a (vo23Tg) transgene. The GFP-Lhfpl5a bundle and FM 4–64 images are from the same NM for each genotype. (K) Quantification of hair cell number in L1, MI1, and O2 neuromasts from 5 dpf lhfpl5bvo35 mutants (n = 11) and wild-type siblings (n = 11). The box plots are the same as in (H). Asterisks indicate p < 0.001 (∗∗∗) by Welch’s t-test. Scale bars = 5 μm, applies to (D–G,I,J); 2 μm in (I,J) insets.
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