- Title
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HOMER2, a Stereociliary Scaffolding Protein, Is Essential for Normal Hearing in Humans and Mice
- Authors
- Azaiez, H., Decker, A.R., Booth, K.T., Simpson, A.C., Shearer, A.E., Huygen, P.L., Bu, F., Hildebrand, M.S., Ranum, P.T., Shibata, S.B., Turner, A., Zhang, Y., Kimberling, W.J., Cornell, R.A., Smith, R.J.
- Source
- Full text @ PLoS Genet.
Over-expression of wtRNA and P185RNA in zebrafish embryos. (A) Otic capsule morphology in non-injected (i, n = 26), wtRNA-injected (ii, n = 26) and P185RNA-injected (iii, n = 27) embryos at 72 hpf. Scale bar represents 50µm. Embryos are mounted anterior to the left. (B) Comparison of otic capsule morphology between WT and mutant RNA injected zebrafish shows reduced inner ear size in mutant RNA injected larvae (p<0.001). (C) Hair cell kinocilia in P185RNA-injected larvae (iii) show fewer kinocilia as compared to non-injected (i) and wtRNA-injected larvae (ii). Scale bar represents 5µm. (D) Kinocilia count per neuromast comparison between wtRNA-injected (n = 56) and P185RNA-injected (n = 55). All data are presented as the mean ± SEM and are derived from at least triplicates (*P < 0.05 as compared with controls considered significant). |
Knock down of homer2 in zebrafish embryos. (A) RT-PCR demonstrating knock down of homer2 transcript using two morpholinos: MO e1i1 and MO i3e4. (B) Representative morphology of otic capsules in zebrafish embryos at 72 hpf either injected with a control MO or MO e1i1. (C) Otic capsule area was measured in 19 and 30 animals in the control and MO groups, respectively. There was no statistically significant difference between groups. Scale bar represents 50µm.PHENOTYPE:
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