ZFIN ID: ZDB-FIG-200829-83
Zhang et al., 2020 - THOC1 deficiency leads to late-onset nonsyndromic hearing loss through p53-mediated hair cell apoptosis. PLoS Genetics   16:e1008953 Full text @ PLoS Genet.
Knockdown Reagent:
Observed In:
Stage Range: Protruding-mouth to Day 4

Fig 4 The THOC1 mutation (p. L183V) impaired its function in hair cell formation.

(a) The diagram shows the targeting site of thoc1 splice blocking morpholino and the RT-PCR primers design for validating the knockdown results. The wild type mature transcripts indicate the natural splicing product of thoc1 mRNA. The splicing MO mature transcripts indicate the abnormal splicing product of thoc1mRNA with Exon3 deletion caused by morpholino injection. (b) The agarose gel electrophoresis image shows the 61bp Exon3 deletion. (c) Confocal microscopic imaging analysis of the hair cells in control, thoc1-MO, thoc1 -MO + hThoc1 mRNA, and thoc1-MO + hThoc1 (p. L183V) mRNA Tg(pou4f3:gap43-GFP) at 3 dpf and 4 dpf. (d) Statistical analysis of the total hair cell number in trunk of control (n = 15), control-MO (n = 15), thoc1-MO (n = 15), thoc1-MO + zthoc1 mRNA (n = 15), thoc1-MO + hThoc1 mRNA (n = 15), and thoc1-MO + hThoc1 (p. L183V) mRNA at 4 dpf (n = 15). One-way ANOVA, ****, p<0.0001.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
s356tTg + MO1-thoc1 standard conditions Protruding-mouth neuromast hair cell decreased amount, abnormal
Protruding-mouth neuromast has fewer parts of type neuromast hair cell, abnormal
Day 4 neuromast hair cell decreased amount, abnormal
Day 4 neuromast has fewer parts of type neuromast hair cell, abnormal
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