Literature DB >> 12057196

The Ig-like structure of the C-terminal domain of lamin A/C, mutated in muscular dystrophies, cardiomyopathy, and partial lipodystrophy.

Isabelle Krimm1, Cecilia Ostlund, Bernard Gilquin, Joël Couprie, Paul Hossenlopp, Jean-Paul Mornon, Gisèle Bonne, Jean-Claude Courvalin, Howard J Worman, Sophie Zinn-Justin.   

Abstract

Lamins are nuclear intermediate filaments that, together with lamin-associated proteins, maintain nuclear shape and provide a structural support for chromosomes and replicating DNA. We have determined the solution structure of the human lamin A/C C-terminal globular domain which contains specific mutations causing four different heritable diseases. This domain encompasses residues 430-545 and adopts an Ig-like fold of type s. We have also characterized by NMR and circular dichroism the structure and thermostability of three mutants, R453W and R482W/Q, corresponding to "hot spots" causing Emery-Dreifuss muscular dystrophy and Dunnigan-type lipodystrophy, respectively. Our structure determination and mutant analyses clearly show that the consequences of the mutations causing muscle-specific diseases or lipodystrophy are different at the molecular level.

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Year:  2002        PMID: 12057196     DOI: 10.1016/s0969-2126(02)00777-3

Source DB:  PubMed          Journal:  Structure        ISSN: 0969-2126            Impact factor:   5.006


  90 in total

1.  Structure and stability of the lamin A tail domain and HGPS mutant.

Authors:  Zhao Qin; Agnieszka Kalinowski; Kris Noel Dahl; Markus J Buehler
Journal:  J Struct Biol       Date:  2011-05-24       Impact factor: 2.867

Review 2.  Lamins at a glance.

Authors:  Chin Yee Ho; Jan Lammerding
Journal:  J Cell Sci       Date:  2012-05-01       Impact factor: 5.285

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.  Nuclear lamin-A scales with tissue stiffness and enhances matrix-directed differentiation.

Authors:  Joe Swift; Irena L Ivanovska; Amnon Buxboim; Takamasa Harada; P C Dave P Dingal; Joel Pinter; J David Pajerowski; Kyle R Spinler; Jae-Won Shin; Manorama Tewari; Florian Rehfeldt; David W Speicher; Dennis E Discher
Journal:  Science       Date:  2013-08-30       Impact factor: 47.728

Review 6.  Delineating the role of alterations in lipid metabolism to the pathogenesis of inherited skeletal and cardiac muscle disorders: Thematic Review Series: Genetics of Human Lipid Diseases.

Authors:  Harjot K Saini-Chohan; Ryan W Mitchell; Frédéric M Vaz; Teresa Zelinski; Grant M Hatch
Journal:  J Lipid Res       Date:  2011-11-07       Impact factor: 5.922

7.  A new LMNA mutation causing limb girdle muscular dystrophy 1B.

Authors:  Simone Spuler; Christian Geier; Karl Josef Osterziel; Matthias Gutberlet; Janine Genschel; Thomas-Nicolas Lehmann; Sophie Zinn-Justin; Bernard Gilquin; Hartmut Schmidt
Journal:  J Neurol       Date:  2005-03-29       Impact factor: 4.849

8.  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

9.  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 10.  Lamins and Lamin-Associated Proteins in Gastrointestinal Health and Disease.

Authors:  Graham F Brady; Raymond Kwan; Juliana Bragazzi Cunha; Jared S Elenbaas; M Bishr Omary
Journal:  Gastroenterology       Date:  2018-03-13       Impact factor: 22.682

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