(A–D) The csr, nco, and vns mutant phenotypes fail to form otoliths within the inner ear. However, semicircular canalformation appears to be normal. (A′–D′) All mutants fail to inflate their swim bladders, which is lethal. Imaged at 5 days post fertilization (dpf). Magnification 6.3×. (*) indicates swim bladder.

Aberrant expression of proteins involved in otolithdevelopment in csr and nco. (A) Schematic of anterior macula (AM) tethered to otolith at 27 hpf. (B) In WT, Otoconin-90 (Oc90) is expressed within the mineralized otolith, which is situated atop the otolithic membrane (Otogelin, or Otog), at 27 hpf. Scale bar = 5 μm. (C–D) Oc90 has diffuse expression within the otocyst of csr and nco. In csr and nco, Otog is localized near the apical surface of hair cells. (E–F) Expression showing hair cells in WT and nco larvae at 5dpf. Scale bar = 25 μm. (G) Quantification of hair cell numbers in the posterior and anterior macula of WT and nco(n = 4).

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