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The Ig-like structure of the C-terminal domain of lamin A/C, mutated in muscular dystrophies, cardiomyopathy, and partial lipodystrophy.

Authors :
Krimm I
Ostlund C
Gilquin B
Couprie J
Hossenlopp P
Mornon JP
Bonne G
Courvalin JC
Worman HJ
Zinn-Justin S
Source :
Structure (London, England : 1993) [Structure] 2002 Jun; Vol. 10 (6), pp. 811-23.
Publication Year :
2002

Abstract

Lamins are nuclear intermediate filaments that, together with lamin-associated proteins, maintain nuclear shape and provide a structural support for chromosomes and replicating DNA. We have determined the solution structure of the human lamin A/C C-terminal globular domain which contains specific mutations causing four different heritable diseases. This domain encompasses residues 430-545 and adopts an Ig-like fold of type s. We have also characterized by NMR and circular dichroism the structure and thermostability of three mutants, R453W and R482W/Q, corresponding to "hot spots" causing Emery-Dreifuss muscular dystrophy and Dunnigan-type lipodystrophy, respectively. Our structure determination and mutant analyses clearly show that the consequences of the mutations causing muscle-specific diseases or lipodystrophy are different at the molecular level.

Details

Language :
English
ISSN :
0969-2126
Volume :
10
Issue :
6
Database :
MEDLINE
Journal :
Structure (London, England : 1993)
Publication Type :
Academic Journal
Accession number :
12057196
Full Text :
https://doi.org/10.1016/s0969-2126(02)00777-3