| Literature DB >> 12057196 |
Isabelle Krimm1, Cecilia Ostlund, Bernard Gilquin, Joël Couprie, Paul Hossenlopp, Jean-Paul Mornon, Gisèle Bonne, Jean-Claude Courvalin, Howard J Worman, Sophie Zinn-Justin.
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.Entities:
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Year: 2002 PMID: 12057196 DOI: 10.1016/s0969-2126(02)00777-3
Source DB: PubMed Journal: Structure ISSN: 0969-2126 Impact factor: 5.006