Work

Comparative synteny cloning of zebrafish you-too: mutations in the Hedgehog target gli2 affect ventral forebrain patterning

Year: 1999

Type: preprint

Abstract: Zebrafish you-too (yot) mutations interfere with Hedgehog (Hh) signaling during embryogenesis. Using a comparative synteny approach, we isolated yot as a zinc finger transcription factor homologous to the Hh target gli2. Two alleles of yot contain nonsense mutations resulting in carboxy-terminally truncated proteins. In addition to causing defects in midline development, muscle differentiation, and retinal axon guidance, yot mutations disrupt anterior pituitary and ventral forebrain differentiation. yot mutations also cause ectopic lens formation in the ventral diencephalon. These findings reveal that truncated zebrafish Gli2 proteins interfere with Hh signaling necessary for differentiation and axon guidance in the ventral forebrain.

Source: Genes & Development

DOI: 10.1101/gad.13.4.388

Authors: R. O. Karlstrom, W. S. Talbot, A. F. Schier

Institution: New York University

Language: English

Topic: Hedgehog Signaling Pathway Studies

Subfield: Molecular Biology

Field: Biochemistry, Genetics and Molecular Biology

Domain: Life Sciences

Open Access status: bronze

Funders: National Institutes of Health, National Institute of Neurological Disorders and Stroke, National Center for Research Resources

Awards: Functional Anaylsis of the Zebrafish Genome, MUTATIONS DISRUPTING AXON GUIDANCE IN ZEBRAFISH