Document Detail


Hutchinson-Gilford progeria mutant lamin A primarily targets human vascular cells as detected by an anti-Lamin A G608G antibody.
MedLine Citation:
PMID:  16461887     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Hutchinson-Gilford progeria syndrome (HGPS; Online Mendelian Inheritance in Man accession no. 176670) is a rare disorder that is characterized by segmental premature aging and death between 7 and 20 years of age from severe premature atherosclerosis. Mutations in the LMNA gene are responsible for this syndrome. Approximately 80% of HGPS cases are caused by a G608 (GGC-->GGT) mutation within exon 11 of LMNA, which elicits a deletion of 50 aa near the C terminus of prelamin A. In this article, we present evidence that the mutant lamin A (progerin) accumulates in the nucleus in a cellular age-dependent manner. In human HGPS fibroblast cultures, we observed, concomitantly to nuclear progerin accumulation, severe nuclear envelope deformations and invaginations preventable by farnesyltransferase inhibition. Nuclear alterations affect cell-cycle progression and cell migration and elicit premature senescence. Strikingly, skin biopsy sections from a subject with HGPS showed that the truncated lamin A accumulates primarily in the nuclei of vascular cells. This finding suggests that accumulation of progerin is directly involved in vascular disease in progeria.
Authors:
Dayle McClintock; Leslie B Gordon; Karima Djabali
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural     Date:  2006-02-06
Journal Detail:
Title:  Proceedings of the National Academy of Sciences of the United States of America     Volume:  103     ISSN:  0027-8424     ISO Abbreviation:  Proc. Natl. Acad. Sci. U.S.A.     Publication Date:  2006 Feb 
Date Detail:
Created Date:  2006-02-15     Completed Date:  2006-04-07     Revised Date:  2009-11-18    
Medline Journal Info:
Nlm Unique ID:  7505876     Medline TA:  Proc Natl Acad Sci U S A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  2154-9     Citation Subset:  IM    
Affiliation:
Department of Dermatology, College of Physicians and Surgeons, Columbia University, New York, NY 10032, USA.
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MeSH Terms
Descriptor/Qualifier:
Age Factors
Aging, Premature / genetics
Antibodies
Atherosclerosis / genetics
Cell Movement
Cell Nucleus / chemistry*,  metabolism
Cell Proliferation
Cells, Cultured
Endothelial Cells / chemistry,  metabolism
Enzyme Inhibitors / pharmacology
Farnesyltranstransferase / antagonists & inhibitors
Fibroblasts / chemistry,  metabolism
Humans
Lamin Type A / analysis*,  genetics*,  metabolism
Mutation
Progeria / genetics,  metabolism*,  pathology
Skin / pathology
Grant Support
ID/Acronym/Agency:
K01AR048594/AR/NIAMS NIH HHS; R01AG025302/AG/NIA NIH HHS
Chemical
Reg. No./Substance:
0/Antibodies; 0/Enzyme Inhibitors; 0/Lamin Type A; EC 2.5.1.29/Farnesyltranstransferase
Comments/Corrections

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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