Fig. 4

Defective neuromast hair cells of L-PLL in zebrafish. A: 4-Di-2-ASP (green) labels neuromasts at 5 dpf. Exposure of a MC larva to 4-Di-2-ASP labels neuromasts (green). Labeling is absent in an MO larva. Coinjection of wild-type RNA coding for wild-type human TMEM132E mostly rescues a MO larva, restoring labeling in most neuromasts. Coinjection of MT RNA barely rescues a MO larva. MT, mutant. Bars, 200 µm. B: Histogram of neuromast numbers of both sides of L-PLL at 5 dpf. C: Scanning electron micrograph of a neuromast from the L-PLL. One midtail neuromast from the PLL of a MC larva at 5 dpf shows 12-long kinocilia emerging from short clusters of stereocilia and the pair of semilunar periderm cells surrounds each neuromast, which are highlighted with yellow dotted line in C (MC). In the MO larvae, the center of a neuromast displays fewer and shorter kinocilia and stereocilia. In addition, the semilunar periderm cells appear distorted.