Fig. 2

Otolith crystallization defects in cep290 mutant homozygotes. (A) Example scanning electron microscopy images of wild-type and cep290^sa1383 otoliths at 18 mo (Scale bar, 300 μm). Enlargements in Bottom panels (Scale bar, 150 μm) (n = 3). (B) Side views of otic vesicles in live wild-type and cep290^sa1383 homozygous embryos at 36 hpf. In the posterior ear, in the mutant example, two closely apposed otoliths are seen, where one is expected. Anterior is Left, dorsal Up, arrowheads indicate otoliths. (C) Frequency of otolith defects at 36 hpf. P < 0.001, χ² test. (D) Typical example of cobalt nitrate staining of utricular otoliths in wild-type and cep290^sa1383 mutants (n = 20).