title: Fragile skeletal muscle attachments in dystrophic mutants of Caenorhabditis elegans: isolation and characterization of the mua genes.
abstract: Over 30 Caenorhabditis elegans mutants were identified with normal muscle differentiation and initial locomotion followed by catastrophic detachment of skeletal muscles from the body wall. Reducing the strength of muscle contraction in these mutants with a myosin gene mutation suppresses muscle detachment. These dystrophic mutants identify a novel class of genes required for growth and maintenance of functional muscle attachments, not exceptional alleles of genes required for muscle differentiation and contractility. Nine new genes, named mua, and two previously published loci, unc-23 and vab-10, cause fragile musscle attachments. The primary sites of muscle detachment, including the plane of tissue separation, are characteristic for each gene. We suggest these genes identify feedback mechanisms whereby local strain regulates the extent of myofibril contraction and the placement of new muscle attachments in functioning muscles. Finally, we draw some comparisons to vertebrate skin fragility diseases and muscular dystrophies.
authors.affiliation: Department of Biology, Johns Hopkins University, Baltimore MD 21218, USA. jplenef@uoft02.utoledo.edu
authors.forename: J D
authors.initials: JD
authors.lastname: Plenefisch
authors.forename: X
authors.initials: X
authors.lastname: Zhu
authors.forename: E M
authors.initials: EM
authors.lastname: Hedgecock
mesh_terms: D000483:Alleles; D000818:Animals; D017173:Caenorhabditis elegans / Q000235:genetics* / Q000254:growth & development* / Q000502:physiology; D002874:Chromosome Mapping; D005246:Feedback; D005260:Female; D018628:Gene Dosage; D017238:Genes, Helminth; D017930:Genes, Reporter; D015801:Helminth Proteins / Q000235:genetics / Q000502:physiology; D009119:Muscle Contraction / Q000235:genetics / Q000502:physiology; D024510:Muscle Development; D009124:Muscle Proteins / Q000235:genetics / Q000502:physiology; D018482:Muscle, Skeletal / Q000254:growth & development*; D009137:Muscular Dystrophy, Animal / Q000235:genetics / Q000473:pathology / Q000503:physiopathology; D009154:Mutation; D010641:Phenotype
chemical_list: D015801:Helminth Proteins; D009124:Muscle Proteins