Abstract
The UDP-N-acetylglucosamine 2-epimerase/N-acetylmannosamine kinase (GNE) gene is the causative gene for autosomal-recessive hereditary inclusion-body myopathy (h-IBM). Two sisters affected with autosomal-recessive h-IBM were shown to be compound heterozygous for two novel GNE mutations: a large deletion involving exons 1-9, and a R162C amino acid change in the epimerase domain. This is the first deletion event observed in a GNE allele and expands the molecular pathogenesis of autosomal-recessive h-IBM.
Copyright 2003 Wiley Periodicals, Inc.
MeSH terms
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Actin Cytoskeleton / pathology
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Adult
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Blotting, Southern
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DNA / genetics
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Exons / genetics
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Female
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Gait Disorders, Neurologic / etiology
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Gene Deletion*
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Genes, Recessive / genetics
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Heterozygote
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Humans
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Microscopy, Electron
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Muscle, Skeletal / pathology
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Mutation, Missense / genetics*
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Mutation, Missense / physiology
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Myositis, Inclusion Body / genetics*
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Myositis, Inclusion Body / pathology
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Phosphotransferases (Alcohol Group Acceptor) / genetics*
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Reverse Transcriptase Polymerase Chain Reaction
Substances
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DNA
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Phosphotransferases (Alcohol Group Acceptor)
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N-acylmannosamine kinase
Associated data
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OMIM/147420
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OMIM/269921
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OMIM/600737
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OMIM/605637
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OMIM/605820