Literature DB >> 11792809

Properties of lamin A mutants found in Emery-Dreifuss muscular dystrophy, cardiomyopathy and Dunnigan-type partial lipodystrophy.

C Ostlund1, G Bonne, K Schwartz, H J Worman.   

Abstract

Autosomal dominant Emery-Dreifuss muscular dystrophy is caused by mutations in the LMNA gene, which encodes lamin A and lamin C. Mutations in this gene also give rise to limb girdle muscular dystrophy type 1B, dilated cardiomyopathy with atrioventricular conduction defect and Dunnigan-type partial lipodystrophy. The properties of the mutant lamins that cause muscular dystrophy, lipodystrophy and dilated cardiomyopathy are not known. We transfected C2C12 myoblasts with cDNA encoding wild-type lamin A and 15 mutant forms found in patients affected by these diseases. Immunofluorescence microscopy showed that four mutants, N195K, E358K, M371K and R386K, could have a dramatically aberrant localization, with decreased nuclear rim staining and formation of intranuclear foci. The distributions of endogenous lamin A/C, lamin B1 and lamin B2 were also altered in cells expressing these four mutants and three of them caused a loss of emerin from the nuclear envelope. In the yeast two-hybrid assay, the 15 lamin A mutants studied interacted with themselves and with wild-type lamin A and lamin B1. Pulse-chase experiments showed no decrease in the stability of several representative lamin A mutants compared with wild-type. These results indicate that some lamin A mutants causing disease can be aberrantly localized, partially disrupt the endogenous lamina and alter emerin localization, whereas others localize normally in transfected cells.

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Year:  2001        PMID: 11792809     DOI: 10.1242/jcs.114.24.4435

Source DB:  PubMed          Journal:  J Cell Sci        ISSN: 0021-9533            Impact factor:   5.285


  74 in total

1.  Expression of a mutant lamin A that causes Emery-Dreifuss muscular dystrophy inhibits in vitro differentiation of C2C12 myoblasts.

Authors:  Catherine Favreau; Dominique Higuet; Jean-Claude Courvalin; Brigitte Buendia
Journal:  Mol Cell Biol       Date:  2004-02       Impact factor: 4.272

Review 2.  How do mutations in lamins A and C cause disease?

Authors:  Howard J Worman; Jean-Claude Courvalin
Journal:  J Clin Invest       Date:  2004-02       Impact factor: 14.808

Review 3.  Nuclear lamins.

Authors:  Thomas Dechat; Stephen A Adam; Pekka Taimen; Takeshi Shimi; Robert D Goldman
Journal:  Cold Spring Harb Perspect Biol       Date:  2010-09-08       Impact factor: 10.005

Review 4.  Inner nuclear membrane proteins: impact on human disease.

Authors:  Iván Méndez-López; Howard J Worman
Journal:  Chromosoma       Date:  2012-02-04       Impact factor: 4.316

5.  Lamin C and chromatin organization in Drosophila.

Authors:  B V Gurudatta; L S Shashidhara; Veena K Parnaik
Journal:  J Genet       Date:  2010-04       Impact factor: 1.166

6.  Functions and dysfunctions of the nuclear lamin Ig-fold domain in nuclear assembly, growth, and Emery-Dreifuss muscular dystrophy.

Authors:  Dale K Shumaker; Reynold I Lopez-Soler; Stephen A Adam; Harald Herrmann; Robert D Moir; Timothy P Spann; Robert D Goldman
Journal:  Proc Natl Acad Sci U S A       Date:  2005-10-14       Impact factor: 11.205

Review 7.  Emery-Dreifuss muscular dystrophy.

Authors:  Antoine Muchir; Howard J Worman
Journal:  Curr Neurol Neurosci Rep       Date:  2007-01       Impact factor: 5.081

Review 8.  Laminopathies: multiple disorders arising from defects in nuclear architecture.

Authors:  Veena K Parnaik; Kaliyaperumal Manju
Journal:  J Biosci       Date:  2006-09       Impact factor: 1.826

9.  Nuclear Lamin Protein C Is Linked to Lineage-Specific, Whole-Cell Mechanical Properties.

Authors:  Rafael D González-Cruz; Jessica S Sadick; Vera C Fonseca; Eric M Darling
Journal:  Cell Mol Bioeng       Date:  2018-01-16       Impact factor: 2.321

10.  In vivo and in vitro examination of the functional significances of novel lamin gene mutations in heart failure patients.

Authors:  N Sylvius; Z T Bilinska; J P Veinot; A Fidzianska; P M Bolongo; S Poon; P McKeown; R A Davies; K-L Chan; A S L Tang; S Dyack; J Grzybowski; W Ruzyllo; H McBride; F Tesson
Journal:  J Med Genet       Date:  2005-08       Impact factor: 6.318

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