Literature DB >> 15372542

Nuclear envelope alterations in fibroblasts from patients with muscular dystrophy, cardiomyopathy, and partial lipodystrophy carrying lamin A/C gene mutations.

A Muchir1, J Medioni, M Laluc, C Massart, T Arimura, A J van der Kooi, I Desguerre, M Mayer, X Ferrer, S Briault, M Hirano, H J Worman, A Mallet, M Wehnert, K Schwartz, G Bonne.   

Abstract

Mutations in LMNA, the gene that encodes nuclear lamins A and C, cause up to eight different diseases collectively referred to as "laminopathies." These diseases affect striated muscle, adipose tissue, peripheral nerve, and bone, or cause features of premature aging. We investigated the consequences of LMNA mutations on nuclear architecture in skin fibroblasts from 13 patients with different laminopathies. Western-blotting showed that none of the mutations examined led to a decrease in cellular levels of lamin A or C. Regardless of the disease, we observed honeycomb nuclear structures and nuclear envelope blebs in cells examined by immunofluorescence microscopy. Concentrated foci of lamin A/C in the nucleoplasm were also observed. Only mutations in the head and tail domains of lamins A and C significantly altered the nuclear architecture of patient fibroblasts. These results confirm that mutations in lamins A and C may lead to a weakening of a structural support network in the nuclear envelope in fibroblasts and that nuclear architecture changes depend upon the location of the mutation in different domains of lamin A/C.

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Year:  2004        PMID: 15372542     DOI: 10.1002/mus.20122

Source DB:  PubMed          Journal:  Muscle Nerve        ISSN: 0148-639X            Impact factor:   3.217


  81 in total

1.  LINCing lamin B2 to neuronal migration: growing evidence for cell-specific roles of B-type lamins.

Authors:  Catherine Coffinier; Loren G Fong; Stephen G Young
Journal:  Nucleus       Date:  2010 Sep-Oct       Impact factor: 4.197

2.  Altering lamina assembly reveals lamina-dependent and -independent functions for A-type lamins.

Authors:  Monika Zwerger; Heidi Roschitzki-Voser; Reto Zbinden; Celine Denais; Harald Herrmann; Jan Lammerding; Markus G Grütter; Ohad Medalia
Journal:  J Cell Sci       Date:  2015-08-14       Impact factor: 5.285

Review 3.  Emery-Dreifuss muscular dystrophy.

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

Review 4.  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

5.  Proteasome-mediated degradation of integral inner nuclear membrane protein emerin in fibroblasts lacking A-type lamins.

Authors:  Antoine Muchir; Catherine Massart; Baziel G van Engelen; Martin Lammens; Gisèle Bonne; Howard J Worman
Journal:  Biochem Biophys Res Commun       Date:  2006-11-03       Impact factor: 3.575

Review 6.  Nuclear mechanics in disease.

Authors:  Monika Zwerger; Chin Yee Ho; Jan Lammerding
Journal:  Annu Rev Biomed Eng       Date:  2011-08-15       Impact factor: 9.590

7.  BioID-based Identification of Skp Cullin F-box (SCF)β-TrCP1/2 E3 Ligase Substrates.

Authors:  Etienne Coyaud; Monika Mis; Estelle M N Laurent; Wade H Dunham; Amber L Couzens; Melanie Robitaille; Anne-Claude Gingras; Stephane Angers; Brian Raught
Journal:  Mol Cell Proteomics       Date:  2015-04-21       Impact factor: 5.911

8.  Skin deep: what can the study of dermal fibroblasts teach us about dilated cardiomyopathy?

Authors:  Brian C Jensen
Journal:  J Mol Cell Cardiol       Date:  2009-12-11       Impact factor: 5.000

9.  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

10.  Myopathic lamin mutations impair nuclear stability in cells and tissue and disrupt nucleo-cytoskeletal coupling.

Authors:  Monika Zwerger; Diana E Jaalouk; Maria L Lombardi; Philipp Isermann; Monika Mauermann; George Dialynas; Harald Herrmann; Lori L Wallrath; Jan Lammerding
Journal:  Hum Mol Genet       Date:  2013-02-19       Impact factor: 6.150

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