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Occipital horn syndrome and a mild Menkes phenotype associated with splice site mutations at the MNK locus.
- Source :
-
Nature genetics [Nat Genet] 1994 Oct; Vol. 8 (2), pp. 195-202. - Publication Year :
- 1994
-
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.
- Subjects :
- Adenosine Triphosphatases chemistry
Adolescent
Animals
Base Sequence
Cells, Cultured
Ceruloplasmin analysis
Copper blood
Copper-Transporting ATPases
DNA Mutational Analysis
Dihydroxyphenylalanine blood
Dihydroxyphenylalanine cerebrospinal fluid
Ehlers-Danlos Syndrome blood
Ehlers-Danlos Syndrome cerebrospinal fluid
Ehlers-Danlos Syndrome classification
Exons
Female
Fibroblasts metabolism
Humans
Male
Menkes Kinky Hair Syndrome blood
Menkes Kinky Hair Syndrome cerebrospinal fluid
Methoxyhydroxyphenylglycol analogs & derivatives
Methoxyhydroxyphenylglycol blood
Methoxyhydroxyphenylglycol cerebrospinal fluid
Mice
Mice, Neurologic Mutants
Molecular Sequence Data
Pedigree
Phenotype
Polymerase Chain Reaction
Sequence Homology, Amino Acid
Species Specificity
Terminator Regions, Genetic
Adenosine Triphosphatases genetics
Carrier Proteins genetics
Cation Transport Proteins
Ehlers-Danlos Syndrome genetics
Menkes Kinky Hair Syndrome genetics
Occipital Bone abnormalities
Point Mutation
RNA Splicing
Recombinant Fusion Proteins
Subjects
Details
- Language :
- English
- ISSN :
- 1061-4036
- Volume :
- 8
- Issue :
- 2
- Database :
- MEDLINE
- Journal :
- Nature genetics
- Publication Type :
- Academic Journal
- Accession number :
- 7842019
- Full Text :
- https://doi.org/10.1038/ng1094-195