The Developmental Genomics Section has been using a combination of zebrafish genetics and molecular embryology to study ear and hearing development. We have focused our attention on the study of several lines isolated in a screen for embryonically lethal mutations in zebrafish. Two of these mutations have been of particular interest. The first is a mutation in the forkhead related transcription factor Foxi1. This mutation has defects in both otic placode formation and jaw development. We are studying the mechanism of function for this transcription factor and are beginning to create a connection between sensory placode development and the formation of craniofacial elements. A second mutation that we are studying involves the disruption of a tight junction protein called claudinJ. Mutations in this gene cause deafness and vestibular defects in zebrafish embryos. We are trying to determine how this occurs. Another focus of the lab is to develop tools for studying gene expression on a genome wide scale. We have been directly involved in the development of an oligo-based microarray for zebrafish genes. We are also working on developing a frozen bank of mapped zebrafish insertional mutations allowing for the isolation of a specific gene disruption.

Agency
National Institute of Health (NIH)
Institute
National Human Genome Research Institute (NHGRI)
Type
Intramural Research (Z01)
Project #
1Z01HG000183-02
Application #
6681683
Study Section
(GTB)
Project Start
Project End
Budget Start
Budget End
Support Year
2
Fiscal Year
2002
Total Cost
Indirect Cost
Name
Human Genome Research
Department
Type
DUNS #
City
State
Country
United States
Zip Code
Liang, Jin; Burgess, Shawn M (2009) Gross and fine dissection of inner ear sensory epithelia in adult zebrafish (Danio rerio). J Vis Exp :
Gomez, Gustavo A; Veldman, Matthew B; Zhao, Yan et al. (2009) Discovery and characterization of novel vascular and hematopoietic genes downstream of etsrp in zebrafish. PLoS One 4:e4994
Jao, Li-En; Burgess, Shawn M (2009) Production of pseudotyped retrovirus and the generation of proviral transgenic zebrafish. Methods Mol Biol 546:13-30
Wang, Ting; Zeng, Jue; Lowe, Craig B et al. (2007) Species-specific endogenous retroviruses shape the transcriptional network of the human tumor suppressor protein p53. Proc Natl Acad Sci U S A 104:18613-8
Wang, Dongmei; Jao, Li-En; Zheng, Naizhong et al. (2007) Efficient genome-wide mutagenesis of zebrafish genes by retroviral insertions. Proc Natl Acad Sci U S A 104:12428-33
Wu, Xiaolin; Luke, Brian T; Burgess, Shawn M (2006) Redefining the common insertion site. Virology 344:292-5
Antonellis, Anthony; Lee-Lin, Shih-Queen; Wasterlain, Amy et al. (2006) Functional analyses of glycyl-tRNA synthetase mutations suggest a key role for tRNA-charging enzymes in peripheral axons. J Neurosci 26:10397-406
Ahmed, Zubair M; Goodyear, Richard; Riazuddin, Saima et al. (2006) The tip-link antigen, a protein associated with the transduction complex of sensory hair cells, is protocadherin-15. J Neurosci 26:7022-34
Yan, Jizhou; Xu, Lisha; Crawford, Gregory et al. (2006) The forkhead transcription factor FoxI1 remains bound to condensed mitotic chromosomes and stably remodels chromatin structure. Mol Cell Biol 26:155-68
Nakai, Hiroyuki; Wu, Xiaolin; Fuess, Sally et al. (2005) Large-scale molecular characterization of adeno-associated virus vector integration in mouse liver. J Virol 79:3606-14

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