Literature DB >> 11313760

The R482Q lamin A/C mutation that causes lipodystrophy does not prevent nuclear targeting of lamin A in adipocytes or its interaction with emerin.

I Holt1, L Clements, S Manilal, S C Brown, G E Morris.   

Abstract

Most pathogenic missense mutations in the lamin A/C gene identified so far cause autosomal-dominant dilated cardiomyopathy and/or Emery-Dreifuss muscular dystrophy. A few specific mutations, however, cause a disease with remarkably different clinical features: FPLD, or familial partial lipodystrophy (Dunnigan-type), which mainly affects adipose tissue. We have prepared lamin A with a known FPLD mutation (R482Q) by in vitro mutagenesis. Nuclear targeting of lamin A in transfected COS cells, human skeletal muscle cells or mouse adipocyte cell cultures (pre- and post-differentiation) was not detectably affected by the mutation. Quantitative in vitro measurements of lamin A interaction with emerin using a biosensor also showed no effect of the mutation. The results show that the loss of function of R482 in lamin A/C in FPLD does not involve loss of ability to form a nuclear lamina or to interact with the nuclear membrane protein, emerin.

Entities:  

Mesh:

Substances:

Year:  2001        PMID: 11313760     DOI: 10.1038/sj.ejhg.5200609

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  6 in total

Review 1.  Lipoatrophic diabetes and other related syndromes.

Authors:  Elif Arioglu Oral
Journal:  Rev Endocr Metab Disord       Date:  2003-03       Impact factor: 6.514

2.  Proteome of human subcutaneous adipose tissue stromal vascular fraction cells versus mature adipocytes based on DIGE.

Authors:  Indu Kheterpal; Ginger Ku; Liana Coleman; Gang Yu; Andrey A Ptitsyn; Z Elizabeth Floyd; Jeffrey M Gimble
Journal:  J Proteome Res       Date:  2011-03-04       Impact factor: 4.466

3.  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

4.  Hutchinson-Gilford progeria mutant lamin A primarily targets human vascular cells as detected by an anti-Lamin A G608G antibody.

Authors:  Dayle McClintock; Leslie B Gordon; Karima Djabali
Journal:  Proc Natl Acad Sci U S A       Date:  2006-02-06       Impact factor: 11.205

5.  Dermal fibroblasts in Hutchinson-Gilford progeria syndrome with the lamin A G608G mutation have dysmorphic nuclei and are hypersensitive to heat stress.

Authors:  Mauro Paradisi; Dayle McClintock; Revekka L Boguslavsky; Christina Pedicelli; Howard J Worman; Karima Djabali
Journal:  BMC Cell Biol       Date:  2005-06-27       Impact factor: 4.241

6.  The Proteome Signatures of Fibroblasts from Patients with Severe, Intermediate and Mild Spinal Muscular Atrophy Show Limited Overlap.

Authors:  Sharon J Brown; Rachel A Kline; Silvia A Synowsky; Sally L Shirran; Ian Holt; Kelly A Sillence; Peter Claus; Brunhilde Wirth; Thomas M Wishart; Heidi R Fuller
Journal:  Cells       Date:  2022-08-23       Impact factor: 7.666

  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.