Gene expression profiling studies on Caenorhabditis elegans dystrophin mutants dys-1(cx-35) and dys-1(cx18).
The Caenorhabditis elegans genome contains a single dystrophin/utrophin orthologue, dys-1. Point mutations in this gene, dys-1(cx35) and dys-1(cx18), result in truncated proteins. Such mutants offer potentially valuable worm models of human Duchenne muscular dystrophy. We have used microarrays to examine genes expressed differentially between wild-type C. elegans and dys-1 mutants. We found 106 genes (115 probe sets) to be differentially expressed when the two mutants are compared to wild-type worms, 49 of which have been assigned to six functional categories. The main categories of regulated genes in C. elegans are genes encoding intracellular signalling, cell-cell communication, cell-surface, and extracellular matrix proteins; genes in these same categories have been shown by others to be differentially expressed in muscle biopsies of muscular dystrophy patients. The C. elegans model may serve as a convenient vehicle for future genetic and chemical screens to search for new drug targets.
Towers
Paula R
PR
MRC Functional Genetics Unit, Department of Human Anatomy and Genetics, University of Oxford, South Parks Road, Oxford OX1 3QX, UK.
Lescure
Pascal
P
Baban
Dilair
D
Malek
Julie A
JA
Duarte
Jose
J
Jones
Emma
E
Davies
Kay E
KE
Ségalat
Laurent
L
Sattelle
David B
DB
D000818:Animals; D001483:Base Sequence; D017173:Caenorhabditis elegans / Q000235:genetics*; D029742:Caenorhabditis elegans Proteins / Q000235:genetics*; D017931:DNA Primers / Q000235:genetics; D018509:DNA, Helminth / Q000235:genetics; D016189:Dystrophin / Q000235:genetics*; D020869:Gene Expression Profiling; D017238:Genes, Helminth; D006801:Humans; D020388:Muscular Dystrophy, Duchenne / Q000235:genetics; D009154:Mutation; D020411:Oligonucleotide Array Sequence Analysis
D029742:Caenorhabditis elegans Proteins; D017931:DNA Primers; D018509:DNA, Helminth; D016189:Dystrophin