The Usher gene cadherin 23 is expressed in the zebrafish brain and a subset of retinal amacrine cells

摘要: PURPOSE: To characterize the expression pattern of cadherin 23 (cdh23) in zebrafish visual system, and to determine whether cdh23 mutants have retinal defects similar those present human disease Usher syndrome 1D. METHODS: In situ hybridization immunohistochemistry were used zebrafish, evaluate for degeneration. Visual function was assessed by measurement optokinetic response siblings mutants. RESULTS: We detected mRNA multiple nuclei both developing adult central nervous system. retina, expressed a small subset amacrine cells, beginning at 70 h postfertilization continuing through adulthood. No photoreceptors. The cdh23-positive population cells GABAergic. Examination homozygous larvae expressing two different mutant alleles cdh23-cdh23(tc317e) or cdh23(tj264a)-revealed no detectable morphological addition, moving gratings varied contrast spatial frequency normal mutants. CONCLUSIONS: Unlike other vertebrates, is not Instead, GABAergic cells. Moreover, with mutations do exhibit any signs gross degeneration dysfunction. role played likely performed either gene an unidentified splice variant retina that affected above mutations.