Abstract
The protein predicted to be defective in individuals with Fanconi anemia complementation group J (FA-J), FANCJ, is a missing component in the Fanconi anemia pathway of genome maintenance. Here we identify pathogenic mutations in eight individuals with FA-J in the gene encoding the DEAH-box DNA helicase BRIP1, also called FANCJ. This finding is compelling evidence that the Fanconi anemia pathway functions through a direct physical interaction with DNA.
MeSH terms
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Chromosomes, Human, Pair 17*
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DNA-Binding Proteins / deficiency*
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DNA-Binding Proteins / genetics*
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Fanconi Anemia / genetics*
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Fanconi Anemia Complementation Group Proteins
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Genetic Complementation Test
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Humans
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Microsatellite Repeats
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Molecular Sequence Data
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Mutation / genetics*
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RNA Helicases / deficiency*
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RNA Helicases / genetics*
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Sequence Deletion
Substances
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DNA-Binding Proteins
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Fanconi Anemia Complementation Group Proteins
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BRIP1 protein, human
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RNA Helicases
Associated data
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RefSeq/NM_032043
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RefSeq/NP_114432