Literature DB >> 11855819

Characterization of adiposity and metabolism in Lmna-deficient mice.

Dedra A Cutler1, Teresa Sullivan, Bernice Marcus-Samuels, Colin L Stewart, Marc L Reitman.   

Abstract

Dunnigan's Familial Partial Lipodystrophy (FPLD) is an autosomal dominant disease characterized by regional fat loss and insulin resistance. FPLD is caused by mutations in the LMNA gene, which encodes intermediate filaments of the nuclear lamina. Different LMNA mutations cause Emery-Dreifuss muscular dystrophy and/or a dilated cardiomyopathy. It is not known how LMNA mutations cause any of the disease phenotypes. Here we measure physical and metabolic characteristics of Lmna-/- and +/- mice to determine their usefulness as models for FPLD. Lmna-/- mice, which die prematurely of muscular dystrophy, have little fat, but do not show the insulin resistance characteristic of FPLD. Lmna+/- mice, despite treatment with a high fat diet, do not have decreased fat stores or metabolic features of FPLD. We also show, in mice, that Lmna transcripts are expressed at high levels in muscle and adipose tissue, but do not vary by body region or sex. In conclusion, Lmna+/- and -/- mice do not mimic Dunnigan's FPLD, and differential expression of lamins A and C does not appear to contribute to sex- or tissue-specific LMNA phenotypes.

Entities:  

Mesh:

Substances:

Year:  2002        PMID: 11855819     DOI: 10.1006/bbrc.2002.6466

Source DB:  PubMed          Journal:  Biochem Biophys Res Commun        ISSN: 0006-291X            Impact factor:   3.575


  18 in total

Review 1.  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

Review 2.  Mouse models of the laminopathies.

Authors:  Colin L Stewart; Serguei Kozlov; Loren G Fong; Stephen G Young
Journal:  Exp Cell Res       Date:  2007-03-31       Impact factor: 3.905

3.  Post-natal myogenic and adipogenic developmental: defects and metabolic impairment upon loss of A-type lamins.

Authors:  Nard Kubben; Jan Willem Voncken; Gonda Konings; Michel van Weeghel; Maarten Mg van den Hoogenhof; Marion Gijbels; Arie van Erk; Kees Schoonderwoerd; Bianca van den Bosch; Vivian Dahlmans; Chantal Calis; Sander M Houten; Tom Misteli; Yigal M Pinto
Journal:  Nucleus       Date:  2011 May-Jun       Impact factor: 4.197

Review 4.  Diseases of the nuclear envelope.

Authors:  Howard J Worman; Cecilia Ostlund; Yuexia Wang
Journal:  Cold Spring Harb Perspect Biol       Date:  2010-02       Impact factor: 10.005

5.  Pathogenic mutations in genes encoding nuclear envelope proteins and defective nucleocytoplasmic connections.

Authors:  Cecilia Östlund; Wakam Chang; Gregg G Gundersen; Howard J Worman
Journal:  Exp Biol Med (Maywood)       Date:  2019-07-12

6.  Extracellular matrix remodeling and transforming growth factor-β signaling abnormalities induced by lamin A/C variants that cause lipodystrophy.

Authors:  Caroline Le Dour; Wei Wu; Véronique Béréziat; Jacqueline Capeau; Corinne Vigouroux; Howard J Worman
Journal:  J Lipid Res       Date:  2016-11-14       Impact factor: 5.922

Review 7.  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

8.  Dual specificity phosphatase 4 mediates cardiomyopathy caused by lamin A/C (LMNA) gene mutation.

Authors:  Jason C Choi; Wei Wu; Antoine Muchir; Shinichi Iwata; Shunichi Homma; Howard J Worman
Journal:  J Biol Chem       Date:  2012-10-09       Impact factor: 5.157

9.  A mouse model of conditional lipodystrophy.

Authors:  Suyeon Kim; Li-Wen Huang; Kathy J Snow; Vitaly Ablamunits; Muneer G Hasham; Timothy H Young; Angelique C Paulk; Joel E Richardson; Jason P Affourtit; Tali Shalom-Barak; Carol J Bult; Yaacov Barak
Journal:  Proc Natl Acad Sci U S A       Date:  2007-10-05       Impact factor: 11.205

Review 10.  "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

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.