Literature DB >> 12729796

Effects of expressing lamin A mutant protein causing Emery-Dreifuss muscular dystrophy and familial partial lipodystrophy in HeLa cells.

Kim Bechert1, Mariana Lagos-Quintana, Jens Harborth, Klaus Weber, Mary Osborn.   

Abstract

Patients with the autosomal dominant form of Emery-Dreifuss muscular dystrophy (EDMD) or familial partial lipodystrophy (FPLD) have specific mutations in the lamin A gene. Three such point mutations, G465D (FPLD), R482L, (FPLD), or R527P (EDMD), were introduced by site-specific mutagenesis in the C-terminal tail domain of a FLAG-tagged full-length lamin A construct. HeLa cells were transfected with mutant and wild-type constructs. Lamin A accumulated in nuclear aggregates and the number of cells with aggregates increased with time after transfection. At 72 h post transfection 60-80% of cells transfected with the mutant lamin A constructs had aggregates, while only 35% of the cells transfected with wild-type lamin A revealed aggregates. Mutant transfected cells expressed 10-24x, and wild-type transfected cells 20x, the normal levels of lamin A. Lamins C, B1 and B2, Nup153, LAP2, and emerin were recruited into aggregates, resulting in a decrease of these proteins at the nuclear rim. Aggregates were also characterized by electron microscopy and found to be preferentially associated with the inner nuclear membrane. Aggregates from mutant constructs were larger than those formed by the wild-type constructs, both in immunofluorescence and electron microscopy. The combined results suggest that aggregate formation is in part due to overexpression, but that there are also mutant-specific effects.

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Year:  2003        PMID: 12729796     DOI: 10.1016/s0014-4827(03)00104-6

Source DB:  PubMed          Journal:  Exp Cell Res        ISSN: 0014-4827            Impact factor:   3.905


  13 in total

Review 1.  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 2.  Versatility at the nuclear pore complex: lessons learned from the nucleoporin Nup153.

Authors:  Jennifer R Ball; Katharine S Ullman
Journal:  Chromosoma       Date:  2005-11-12       Impact factor: 4.316

Review 3.  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

4.  Biomechanical defects and rescue of cardiomyocytes expressing pathologic nuclear lamins.

Authors:  Erik Laurini; Valentina Martinelli; Thomas Lanzicher; Luca Puzzi; Daniele Borin; Suet Nee Chen; Carlin S Long; Patrice Lee; Luisa Mestroni; Matthew R G Taylor; Orfeo Sbaizero; Sabrina Pricl
Journal:  Cardiovasc Res       Date:  2018-05-01       Impact factor: 10.787

5.  Lamin A N-terminal phosphorylation is associated with myoblast activation: impairment in Emery-Dreifuss muscular dystrophy.

Authors:  V Cenni; P Sabatelli; E Mattioli; S Marmiroli; C Capanni; A Ognibene; S Squarzoni; N M Maraldi; G Bonne; M Columbaro; L Merlini; G Lattanzi
Journal:  J Med Genet       Date:  2005-03       Impact factor: 6.318

Review 6.  "Laminopathies": a wide spectrum of human diseases.

Authors:  Howard J Worman; Gisèle Bonne
Journal:  Exp Cell Res       Date:  2007-03-30       Impact factor: 3.905

7.  Defects in nuclear structure and function promote dilated cardiomyopathy in lamin A/C-deficient mice.

Authors:  Vesna Nikolova; Christiana Leimena; Aisling C McMahon; Ju Chiat Tan; Suchitra Chandar; Dilesh Jogia; Scott H Kesteven; Jan Michalicek; Robyn Otway; Fons Verheyen; Stephen Rainer; Colin L Stewart; David Martin; Michael P Feneley; Diane Fatkin
Journal:  J Clin Invest       Date:  2004-02       Impact factor: 14.808

8.  A Role of Lamin A/C in Preventing Neuromuscular Junction Decline in Mice.

Authors:  Nannan Gao; Kai Zhao; Yu Cao; Xiao Ren; Hongyang Jing; Guanglin Xing; Wen-Cheng Xiong; Lin Mei
Journal:  J Neurosci       Date:  2020-08-17       Impact factor: 6.167

9.  Morphological analysis of 13 LMNA variants identified in a cohort of 324 unrelated patients with idiopathic or familial dilated cardiomyopathy.

Authors:  Jason Cowan; Duanxiang Li; Jorge Gonzalez-Quintana; Ana Morales; Ray E Hershberger
Journal:  Circ Cardiovasc Genet       Date:  2009-11-17

10.  Characterization of lamin mutation phenotypes in Drosophila and comparison to human laminopathies.

Authors:  Andrés Muñoz-Alarcón; Maja Pavlovic; Jasmine Wismar; Bertram Schmitt; Maria Eriksson; Per Kylsten; Mitchell S Dushay
Journal:  PLoS One       Date:  2007-06-13       Impact factor: 3.240

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