ZFIN ID: ZDB-FIG-200116-1
Rissone et al., 2019 - A model for reticular dysgenesis shows impaired sensory organ development and hair cell regeneration linked to cellular stress. Disease models & mechanisms   12(12) Full text @ Dis. Model. Mech.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes:
Fish:
Anatomical Terms:
Stage Range: Protruding-mouth to Day 5
PHENOTYPE:
Fish:
Observed In:
Stage Range: Protruding-mouth to Days 7-13

Fig. 1.

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).

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
ak2 WT control Protruding-mouth otic vesicle ISH
Day 4 otic vesicle ISH
Day 5 otic vesicle ISH
GFP ak2hg14/hg14; s356tTg control Protruding-mouth anterior crista hair cell IFL
Protruding-mouth hair cell anterior macula IFL
Protruding-mouth lateral crista hair cell IFL
Protruding-mouth posterior crista hair cell IFL
Day 4 anterior crista hair cell IFL
Day 4 lateral crista hair cell IFL
Day 4 posterior crista hair cell IFL
s356tTg control Protruding-mouth anterior crista hair cell IFL
Protruding-mouth hair cell anterior macula IFL
Protruding-mouth lateral crista hair cell IFL
Protruding-mouth posterior crista hair cell IFL
Day 4 anterior crista hair cell IFL
Day 4 lateral crista hair cell IFL
Day 4 posterior crista hair cell IFL
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
ak2hg14/hg14 standard conditions Day 4 - Days 7-13 whole organism decreased life span, abnormal
Days 7-13 whole organism dead, abnormal
ak2hg14/hg14; s356tTg control Protruding-mouth auditory receptor cell decreased amount, abnormal
Protruding-mouth hair cell anterior macula decreased amount, abnormal
Day 4 auditory receptor cell decreased amount, abnormal
ak2hg16/hg16 standard conditions Day 4 - Days 7-13 whole organism decreased life span, abnormal
Acknowledgments:
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