Literature DB >> 7842019

Occipital horn syndrome and a mild Menkes phenotype associated with splice site mutations at the MNK locus.

S G Kaler1, L K Gallo, V K Proud, A K Percy, Y Mark, N A Segal, D S Goldstein, C S Holmes, W A Gahl.   

Abstract

We have found mutations in the Menkes disease gene (MNK) which impair, but do not abolish, correct mRNA splicing in patients with less severe clinical phenotypes. In one family, four males aged 2-36 years with a distinctive Menkes variant have a mutation at the +3 position of a splice donor site near the 3' end of the Menkes coding sequence that is associated with exon skipping and a stable mutant transcript. In an unrelated 15-year-old male with typical occipital horn syndrome, a point mutation at the -2 exonic position of a splice donor site in the middle of the gene causes exon-skipping and activation of a cryptic splice acceptor site. In both mutations, maintenance of some normal splicing is demonstrable by RT-PCR, cDNA sequencing and ribonuclease protection.

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Year:  1994        PMID: 7842019     DOI: 10.1038/ng1094-195

Source DB:  PubMed          Journal:  Nat Genet        ISSN: 1061-4036            Impact factor:   38.330


  82 in total

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6.  Functional expression of the Wilson disease protein reveals mislocalization and impaired copper-dependent trafficking of the common H1069Q mutation.

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Review 9.  Copper transporting P-type ATPases and human disease.

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