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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.
- 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.
- Subjects :
- Amino Acid Sequence
Cardiomyopathies metabolism
Circular Dichroism
Conserved Sequence
Immunoglobulins genetics
Lamin Type A metabolism
Lipodystrophy metabolism
Magnetic Resonance Spectroscopy
Molecular Sequence Data
Muscular Dystrophies metabolism
Mutation
Phenotype
Protein Structure, Tertiary
Sequence Alignment
Sequence Analysis, Protein
Cardiomyopathies genetics
Lamin Type A genetics
Lipodystrophy genetics
Muscular Dystrophies genetics
Subjects
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