Using genomewide mutagenesis screens to identify the genes required for neural tube closure in the mouse Journal Article


Authors: Zohn, I. E.; Anderson, K. V.; Niswander, L.
Article Title: Using genomewide mutagenesis screens to identify the genes required for neural tube closure in the mouse
Abstract: BACKGROUND: Neural tube closure is a critical embryological process that requires the coordination of many molecular and cellular events. Only recently has the molecular basis of the cell movements that drive neural tube closure begun to be elucidated. This has been accomplished in part due to the analysis of a growing number of genetically targeted and naturally occurring mouse mutant strains that have neural tube defects (NTDs). Currently there are more than 100 genes that when mutated result in NTDs in the mouse. Yet only approximately 10% of genes in the mouse genome have been mutated and their gross phenotype analyzed, suggesting that only a small percentage of the genes that can cause NTDs have been identified. METHODS: In order to more systematically and fully understand the genetic basis of neural tube closure and to begin to define the molecular pathways that direct this key embryonic event, our laboratories have undertaken a forward genetic screen in mice. From this we hope to gain a better understanding of the regulation of this complex morphogenic processes. CONCLUSIONS: The mouse provides a good model for human neural tube closure, and therefore the information gained from generating novel mouse models of NTDs will help to predict the genes responsible for human NTDs and provide experimental evidence for how they function. © 2005 Wiley-Liss, Inc.
Keywords: signal transduction; review; nonhuman; animals; mice; embryo; sonic hedgehog protein; animal model; mice, mutant strains; embryo development; genome; models, genetic; disease models, animal; genetic screening; nervous system development; mutagenesis; neural tube defect; birth defect; neural tube defects
Journal Title: Birth Defects Research Part A - Clinical and Molecular Teratology
Volume: 73
Issue: 9
ISSN: 1542-0752
Publisher: Wiley Liss  
Date Published: 2005-09-01
Start Page: 583
End Page: 590
Language: English
DOI: 10.1002/bdra.20164
PUBMED: 15971254
PROVIDER: scopus
DOI/URL:
Notes: --- - "Cited By (since 1996): 23" - "Export Date: 24 October 2012" - "CODEN: BDRPB" - "Source: Scopus"
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  1. Irene Zohn
    3 Zohn
  2. Kathryn Anderson
    148 Anderson