Literature DB >> 12748643

A progeroid syndrome in mice is caused by defects in A-type lamins.

Leslie C Mounkes1, Serguei Kozlov, Lidia Hernandez, Teresa Sullivan, Colin L Stewart.   

Abstract

Numerous studies of the underlying causes of ageing have been attempted by examining diseases associated with premature ageing, such as Werner's syndrome and Hutchinson-Gilford progeria syndrome (HGPS). HGPS is a rare genetic disorder resulting in phenotypes suggestive of accelerated ageing, including shortened stature, craniofacial disproportion, very thin skin, alopecia and osteoporosis, with death in the early teens predominantly due to atherosclerosis. However, recent reports suggest that developmental abnormalities may also be important in HGPS. Here we describe the derivation of mice carrying an autosomal recessive mutation in the lamin A gene (Lmna) encoding A-type lamins, major components of the nuclear lamina. Homozygous mice display defects consistent with HGPS, including a marked reduction in growth rate and death by 4 weeks of age. Pathologies in bone, muscle and skin are also consistent with progeria. The Lmna mutation resulted in nuclear morphology defects and decreased lifespan of homozygous fibroblasts, suggesting premature cell death. Here we present a mouse model for progeria that may elucidate mechanisms of ageing and development in certain tissue types, especially those developing from the mesenchymal cell lineage.

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Year:  2003        PMID: 12748643     DOI: 10.1038/nature01631

Source DB:  PubMed          Journal:  Nature        ISSN: 0028-0836            Impact factor:   49.962


  112 in total

Review 1.  A-type lamin complexes and regenerative potential: a step towards understanding laminopathic diseases?

Authors:  Josef Gotzmann; Roland Foisner
Journal:  Histochem Cell Biol       Date:  2005-09-02       Impact factor: 4.304

Review 2.  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

3.  Molecular genetic analysis of the nested Drosophila melanogaster lamin C gene.

Authors:  Sandra R Schulze; Beatrice Curio-Penny; Yuhong Li; Reza A Imani; Lena Rydberg; Pamela K Geyer; Lori L Wallrath
Journal:  Genetics       Date:  2005-06-18       Impact factor: 4.562

Review 4.  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

Review 5.  Adult stem cell maintenance and tissue regeneration in the ageing context: the role for A-type lamins as intrinsic modulators of ageing in adult stem cells and their niches.

Authors:  Vanja Pekovic; Christopher J Hutchison
Journal:  J Anat       Date:  2008-07       Impact factor: 2.610

6.  Role of the nuclear envelope in the pathogenesis of age-related bone loss and osteoporosis.

Authors:  Christopher Vidal; Sandra Bermeo; Diane Fatkin; Gustavo Duque
Journal:  Bonekey Rep       Date:  2012-05-02

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.  Reversal of the cellular phenotype in the premature aging disease Hutchinson-Gilford progeria syndrome.

Authors:  Paola Scaffidi; Tom Misteli
Journal:  Nat Med       Date:  2005-03-06       Impact factor: 53.440

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

Review 10.  The role of lamin A/C in mesenchymal stem cell differentiation.

Authors:  Bo Zhang; Yang Yang; Reziwan Keyimu; Jin Hao; Zhihe Zhao; Rui Ye
Journal:  J Physiol Biochem       Date:  2019-01-31       Impact factor: 4.158

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