ak2 larval expression in the otic vesicle and sensory phenotypes of the zebrafish ak2 alleles. (A) WISH on wild-type zebrafish larvae at different developmental stages. Lateral (3-5 dpf) and dorsal (5 dpf) views. Black arrows point to ak2 expression in the otic vesicles. Scale bars: 200 μm. (B) Survival rates for wild-type, ak2 heterozygous, and ak2 mutant larvae at different dpf. Top and bottom graphs show survival of the progeny from an incross of ak2hg16/+ or ak2hg14/+ heterozygous animals, respectively. Dashed lines indicate the expected ratio following Mendelian inheritance. Numbers above the bars indicate the total number of embryos used for genotyping analysis at each stage. (C,D) Comparison of confocal maximum projections of inner ear regions (dashed circles) from Tg(pou4f3:GAP-GFP) wild type (WT) and ak2hg14 mutants at 3 (C) and 4 (D) dpf. Scale bars: 20 µm. For each set of images, graphs show the comparison of the average number of GFP+ hair cells in the anterior macula (3 dpf) and cristae (3 and 4 dpf) between ak2-deficient animals and controls (WT). a, anterior; ac, anterior crista; am, anterior macula; HCs, hair cells; m, medial; mc, medial crista; NM, neuromast; p, posterior; pc, posterior crista. The error bars indicate s.d. ****P<0.0001 (two-tailed unpaired Student's test).
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