Document Detail

The detached locus encodes Drosophila Dystrophin, which acts with other components of the Dystrophin Associated Protein Complex to influence intercellular signalling in developing wing veins.
MedLine Citation:
PMID:  18093579     Owner:  NLM     Status:  MEDLINE    
Dystrophin and Dystroglycan are the two central components of the multimeric Dystrophin Associated Protein Complex, or DAPC, that is thought to provide a mechanical link between the extracellular matrix and the actin cytoskeleton, disruption of which leads to muscular dystrophy in humans. We present the characterization of the Drosophila 'crossveinless' mutation detached (det), and show that the gene encodes the fly ortholog of Dystrophin. Our genetic analysis shows that, in flies, Dystrophin is a non-essential gene, and the sole overt morphological defect associated with null mutations in the locus is the variable loss of the posterior crossvein that has been described for alleles of det. Null mutations in Drosophila Dystroglycan (Dg) are similarly viable and exhibit this crossvein defect, indicating that both of the central DAPC components have been co-opted for this atypical function of the complex. In the developing wing, the Drosophila DAPC affects the intercellular signalling pathways involved in vein specification. In det and Dg mutant wings, the early BMP signalling that initiates crossvein specification is not maintained, particularly in the pro-vein territories adjacent to the longitudinal veins, and this results in the production of a crossvein fragment in the intervein between the two longitudinal veins. Genetic interaction studies suggest that the DAPC may exert this effect indirectly by down-regulating Notch signalling in pro-vein territories, leading to enhanced BMP signalling in the intervein by diffusion of BMP ligands from the longitudinal veins.
Christina P Christoforou; Claire E Greer; Benjamin R Challoner; Dimitris Charizanos; Robert P Ray
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't     Date:  2007-09-29
Journal Detail:
Title:  Developmental biology     Volume:  313     ISSN:  1095-564X     ISO Abbreviation:  Dev. Biol.     Publication Date:  2008 Jan 
Date Detail:
Created Date:  2008-01-14     Completed Date:  2008-02-26     Revised Date:  2014-02-19    
Medline Journal Info:
Nlm Unique ID:  0372762     Medline TA:  Dev Biol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  519-32     Citation Subset:  IM    
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MeSH Terms
Chromosome Mapping
Drosophila / embryology,  genetics*
Dystroglycans / genetics,  physiology
Dystrophin / genetics*,  metabolism
Dystrophin-Associated Protein Complex / genetics,  metabolism*
Embryo, Nonmammalian
Gene Expression Regulation, Developmental
Genes, Insect*
Models, Biological
Signal Transduction*
Wing / anatomy & histology,  embryology*,  metabolism
beta-Galactosidase / metabolism
Grant Support
G0500916//Medical Research Council
Reg. No./Substance:
0/Dystrophin; 0/Dystrophin-Associated Protein Complex; 146888-27-9/Dystroglycans; EC

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