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PMID: 8490646 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Isolation of a candidate gene for Menkes disease that encodes a potential heavy metal binding protein.

Nature genetics ·Vol. 3 ·No. 1 ·1993-01-00 ·Pages 14-9

Chelly J, Tümer Z, Tønnesen T, Petterson A, Ishikawa-Brush Y, Tommerup N, Horn N, Monaco AP

Abstract

Menkes disease is a lethal-X linked recessive disorder associated with copper metabolism disturbance. We have recently mapped two chromosome breakpoints related to this disease in a 1 megabase yeast artificial chromosome contig at Xq13.3. We now report the construction of a phage contig and the isolation of candidate partial cDNAs for the Menkes disease gene. The candidate gene expresses an 8 kb message in all investigated tissues, and deletions were detected in 16% of 100 unrelated Menkes patients. The deduced partial protein sequence shared the GMTCXXC motif with bacterial metal resistance operons, suggesting a potential heavy metal binding protein. These findings should lead to more accurate prenatal diagnosis of this severe disease and a better understanding of the cellular homeostasis of essential heavy metals.

MeSH Terms
Adenosine Triphosphatases/genetics,metabolism Amino Acid Sequence Base Sequence Blotting, Southern Carrier Proteins/genetics,metabolism Cation Transport Proteins Cells, Cultured Cloning, Molecular Copper-Transporting ATPases DNA Female Humans Male Menkes Kinky Hair Syndrome/genetics Metals/metabolism Molecular Sequence Data Recombinant Fusion Proteins Sequence Homology, Amino Acid X Chromosome
Chemicals
Carrier Proteins Cation Transport Proteins Metals Recombinant Fusion Proteins DNA Adenosine Triphosphatases ATP7A protein, human Copper-Transporting ATPases
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Chelly J
ICRF Laboratories, John Radcliffe Hospital, Headington, Oxford, UK.
Tümer Z
Tønnesen T
Petterson A
Ishikawa-Brush Y
Tommerup N
Horn N
Monaco A P
Article Info
Journal
Nature genetics
Abbr.
Nat Genet
ISSN
1061-4036
Published
1993-01-00
Pages
14-9
Language
English
Region
United States
NLM ID
9216904
Subset
IM
Databases
GENBANK
X69208
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