Zebrafish Models of Human Disease

National Human Genome Research Institute

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Abstract

Background: We wishg to generalize our ability to measure candidate human allele functionality in zebrafish, with the sole requirements being that the gene in question is sufficiently analogous to a zebrafish gene with embryonic activity. For this we have pioneered a new platform, Nanostring, to count and correlate the quantity of injected WT and candidate mutant alleles of human RNAs with the output (activation or repression ) of a signature subset of zebrafish genes. We have applied this to two test scenarios, both in the sonic hedgehog signaling pathway: human SHH alleles and human GLI2 alleles. The results re good and we are preparing manuscripts to report upon this method and our new data.