| Literature DB >> 12490190 |
Catherine Favreau1, Emmanuelle Dubosclard, Cecilia Ostlund, Corinne Vigouroux, Jacqueline Capeau, Manfred Wehnert, Dominique Higuet, Howard J Worman, Jean-Claude Courvalin, Brigitte Buendia.
Abstract
Autosomal dominantly inherited missense mutations in lamins A and C cause familial partial lipodystrophy of the Dunnigan-type (FPLD), and myopathies including Emery-Dreifuss muscular dystrophy (EDMD). While mutations responsible for FPLD are restricted to the carboxyl-terminal tails, those responsible for EDMD are spread throughout the molecules. We observed here the same structural abnormalities in the nuclear envelope and chromatin of fibroblasts from patients with FPLD and EDMD, harboring missense mutations at codons 482 and 453, respectively. Similar nuclear alterations were generated in fibroblasts, myoblasts, and preadipocytes mouse cell lines overexpressing lamin A harboring either of these two mutations. A large variation in sensitivity to lamin A overexpression was observed among the three cell lines, which was correlated with their variable endogenous content in A-type lamins and emerin. The occurrence of nuclear abnormalities was reduced when lamin B1 was coexpressed with mutant lamin A, emphasizing the functional interaction of the two types of lamins. Transfected cells therefore develop similar phenotypes when expressing lamins mutated in the carboxyl-terminal tail at sites responsible for FPLD or EDMD.Entities:
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Year: 2003 PMID: 12490190 DOI: 10.1006/excr.2002.5669
Source DB: PubMed Journal: Exp Cell Res ISSN: 0014-4827 Impact factor: 3.905