UNC-119 homolog required for normal development of the zebrafish nervous system

The UNC‐119 proteins, found in all metazoans examined, are highly conserved at both the sequence and functional levels. In the invertebrates Caenorhabditis elegans and Drosophila melanogaster, unc‐119 genes are expressed pan‐neurally. Loss of function of the unc‐119 gene in C. elegans results in a d...

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Published in:	Genesis (New York, N.Y. : 2000) Vol. 40; no. 4; pp. 223 - 230
Main Authors:	Manning, Angela G., Crawford, Bryan D., Waskiewicz, Andrew J., Pilgrim, David B.
Format:	Journal Article
Language:	English
Published:	Hoboken Wiley Subscription Services, Inc., A Wiley Company 01-12-2004
Subjects:	Amino Acid Sequence Animals axonal branching Body Patterning Caenorhabditis elegans Danio rerio defasciculation Drosophila melanogaster Immunoblotting Molecular Sequence Data Nerve Tissue Proteins - genetics Nervous System - embryology Nervous System - metabolism neural development Radiation Hybrid Mapping Recombinant Fusion Proteins - genetics Recombinant Fusion Proteins - metabolism Sequence Homology, Amino Acid unc-119 zebrafish Zebrafish - embryology Zebrafish - genetics Zebrafish Proteins - genetics
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Summary:	The UNC‐119 proteins, found in all metazoans examined, are highly conserved at both the sequence and functional levels. In the invertebrates Caenorhabditis elegans and Drosophila melanogaster, unc‐119 genes are expressed pan‐neurally. Loss of function of the unc‐119 gene in C. elegans results in a disorganized neural architecture and paralysis. The function of UNC‐119 proteins has been conserved throughout evolution, as transgenic expression of the human UNC119 gene in C. elegans unc‐119 mutants restores a wild‐type phenotype. However, the nature of the conserved molecular function of UNC‐119 proteins is poorly understood. Although unc‐119 genes are expressed throughout the nervous system of the worm and fly, the analysis of these genes in vertebrates has focused on their function in the photoreceptor cells of the retina. Here we report the characterization of an unc‐119 homolog in the zebrafish. The Unc119 protein is expressed in various neural tissues in the developing zebrafish embryo and larva. Morpholino oligonucleotide (MO)‐mediated knockdown of Unc119 protein results in a “curly tail down” phenotype. Examination of neural patterning demonstrates that these “curly tail down” zebrafish experience a constellation of neuronal defects similar to those seen in C. elegans unc‐119 mutants: missing or misplaced cell bodies, process defasciculation, axon pathfinding errors, and aberrant axonal branching. These findings suggest that UNC‐119 proteins may play an important role in the development and/or function of the vertebrate nervous system. genesis 40:223–230, 2004. © 2004 Wiley‐Liss, Inc.
Bibliography:	ark:/67375/WNG-HKQXRB9T-F ArticleID:GENE20089 istex:A432138FA1B3CC9F0DDFA2FC896E8BD8BD69F950 Canadian Institutes of Health Research ObjectType-Article-2 SourceType-Scholarly Journals-1 ObjectType-Feature-1 content type line 23 SourceType-Other Sources-1 content type line 63 ObjectType-Correspondence-1
ISSN:	1526-954X 1526-968X
DOI:	10.1002/gene.20089