RRC ID 28846
著者 Morriss GR, Jaramillo CT, Mikolajczak CM, Duong S, Jaramillo MS, Cripps RM.
タイトル The Drosophila wings apart gene anchors a novel, evolutionarily conserved pathway of neuromuscular development.
ジャーナル Genetics
Abstract wings apart (wap) is a recessive, semilethal gene located on the X chromosome in Drosophila melanogaster, which is required for normal wing-vein patterning. We show that the wap mutation also results in loss of the adult jump muscle. We use complementation mapping and gene-specific RNA interference to localize the wap locus to the proximal X chromosome. We identify the annotated gene CG14614 as the gene affected by the wap mutation, since one wap allele contains a non-sense mutation in CG14614, and a genomic fragment containing only CG14614 rescues the jump-muscle phenotypes of two wap mutant alleles. The wap gene lies centromere-proximal to touch-insensitive larva B and centromere-distal to CG14619, which is tentatively assigned as the gene affected in introverted mutants. In mutant wap animals, founder cell precursors for the jump muscle are specified early in development, but are later lost. Through tissue-specific knockdowns, we demonstrate that wap function is required in both the musculature and the nervous system for normal jump-muscle formation. wap/CG14614 is homologous to vertebrate wdr68, DDB1 and CUL4 associated factor 7, which also are expressed in neuromuscular tissues. Thus, our findings provide insight into mechanisms of neuromuscular development in higher animals and facilitate the understanding of neuromuscular diseases that may result from mis-expression of muscle-specific or neuron-specific genes.
巻・号 195(3)
ページ 927-40
公開日 2013-11-1
DOI 10.1534/genetics.113.154211
PII genetics.113.154211
PMID 24026097
PMC PMC3813874
MeSH Animals Chromosome Mapping Conserved Sequence Drosophila melanogaster / genetics* Drosophila melanogaster / growth & development* Drosophila melanogaster / physiology Evolution, Molecular Female Gene Expression Regulation, Developmental Genes, Insect* Genetic Complementation Test Male Mutation Neuromuscular Junction / genetics* Neuromuscular Junction / growth & development* Neuromuscular Junction / physiology Phenotype RNA Interference Wings, Animal / growth & development
IF 4.015
引用数 4
WOS 分野 GENETICS & HEREDITY
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