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PMID: 10655060 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

LMNA, encoding lamin A/C, is mutated in partial lipodystrophy.

Nature genetics ·Vol. 24 ·No. 2 ·2000-02-00 ·Pages 153-6

Shackleton S, Lloyd DJ, Jackson SN, Evans R, Niermeijer MF, Singh BM, Schmidt H, Brabant G, Kumar S, Durrington PN, Gregory S, O'Rahilly S, Trembath RC

Abstract

The lipodystrophies are a group of disorders characterized by the absence or reduction of subcutaneous adipose tissue. Partial lipodystrophy (PLD; MIM 151660) is an inherited condition in which a regional (trunk and limbs) loss of fat occurs during the peri-pubertal phase. Additionally, variable degrees of resistance to insulin action, together with a hyperlipidaemic state, may occur and simulate the metabolic features commonly associated with predisposition to atherosclerotic disease. The PLD locus has been mapped to chromosome 1q with no evidence of genetic heterogeneity. We, and others, have refined the location to a 5.3-cM interval between markers D1S305 and D1S1600 (refs 5, 6). Through a positional cloning approach we have identified five different missense mutations in LMNA among ten kindreds and three individuals with PLD. The protein product of LMNA is lamin A/C, which is a component of the nuclear envelope. Heterozygous mutations in LMNA have recently been identified in kindreds with the variant form of muscular dystrophy (MD) known as autosomal dominant Emery-Dreifuss MD (EDMD-AD; ref. 7) and dilated cardiomyopathy and conduction-system disease (CMD1A). As LMNA is ubiquitously expressed, the finding of site-specific amino acid substitutions in PLD, EDMD-AD and CMD1A reveals distinct functional domains of the lamin A/C protein required for the maintenance and integrity of different cell types.

MeSH Terms
Amino Acid Sequence Amino Acid Substitution Animals Base Sequence Chromosome Mapping Chromosomes, Human, Pair 1 Female Genetic Markers Heterozygote Humans Lamin Type A Lamins Lipodystrophy/genetics Male Mice Molecular Sequence Data Nuclear Proteins/chemistry,genetics Pedigree Point Mutation Rats Sequence Alignment Sequence Homology, Amino Acid
Chemicals
Genetic Markers Lamin Type A Lamins Nuclear Proteins
Authors & Affiliations
13 authors, click to expand affiliations / ORCID
Shackleton S
Division of Medical Genetics, Departments of Medicine and Genetics, University of Leicester, Leicester, UK.
Lloyd D J
Jackson S N
Evans R
Niermeijer M F
Singh B M
Schmidt H
Brabant G
Kumar S
Durrington P N
Gregory S
O'Rahilly S
Trembath R C
Article Info
Journal
Nature genetics
Abbr.
Nat Genet
ISSN
1061-4036
Published
2000-02-00
Pages
153-6
Language
English
Region
United States
NLM ID
9216904
Subset
IM
Corrections
CommentIn
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