Literature DB >> 31009944

Postnatal development of mice with combined genetic depletions of lamin A/C, emerin and lamina-associated polypeptide 1.

Yuexia Wang1,2, Ji-Yeon Shin1,2, Koki Nakanishi1, Shunichi Homma1, Grace J Kim1, Kurenai Tanji2, Leroy C Joseph1, John P Morrow1, Colin L Stewart3, Willian T Dauer4,5, Howard J Worman1,2.   

Abstract

Mutations in LMNA encoding lamin A/C and EMD encoding emerin cause cardiomyopathy and muscular dystrophy. Lmna null mice develop these disorders and have a lifespan of 7-8 weeks. Emd null mice show no overt pathology and have normal skeletal muscle but with regeneration defects. We generated mice with germline deletions of both Lmna and Emd to determine the effects of combined loss of the encoded proteins. Mice without lamin A/C and emerin are born at the expected Mendelian ratio, are grossly normal at birth but have shorter lifespans than those lacking only lamin A/C. However, there are no major differences between these mice with regards to left ventricular function, heart ultrastructure or electrocardiographic parameters except for slower heart rates in the mice lacking both lamin A/C and emerin. Skeletal muscle is similarly affected in both of these mice. Lmna+/- mice also lacking emerin live to at least 1 year and have no significant differences in growth, heart or skeletal muscle compared to Lmna+/- mice. Deletion of the mouse gene encoding lamina-associated protein 1 leads to prenatal death; however, mice with heterozygous deletion of this gene lacking both lamin A/C and emerin are born at the expected Mendelian ratio but had a shorter lifespan than those only lacking lamin A/C and emerin. These results show that mice with combined deficiencies of three interacting nuclear envelope proteins have normal embryonic development and that early postnatal defects are primarily driven by loss of lamin A/C or lamina-associated polypeptide 1 rather than emerin.
© The Author(s) 2019. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com.

Entities:  

Year:  2019        PMID: 31009944      PMCID: PMC7590221          DOI: 10.1093/hmg/ddz082

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  72 in total

1.  A molecular mechanism underlying the neural-specific defect in torsinA mutant mice.

Authors:  Connie E Kim; Alex Perez; Guy Perkins; Mark H Ellisman; William T Dauer
Journal:  Proc Natl Acad Sci U S A       Date:  2010-05-10       Impact factor: 11.205

2.  Homologies in both primary and secondary structure between nuclear envelope and intermediate filament proteins.

Authors:  F D McKeon; M W Kirschner; D Caput
Journal:  Nature       Date:  1986 Feb 6-12       Impact factor: 49.962

3.  Missense mutations in the rod domain of the lamin A/C gene as causes of dilated cardiomyopathy and conduction-system disease.

Authors:  D Fatkin; C MacRae; T Sasaki; M R Wolff; M Porcu; M Frenneaux; J Atherton; H J Vidaillet; S Spudich; U De Girolami; J G Seidman; C Seidman; F Muntoni; G Müehle; W Johnson; B McDonough
Journal:  N Engl J Med       Date:  1999-12-02       Impact factor: 91.245

4.  Blocking farnesylation of the prelamin A variant in Hutchinson-Gilford progeria syndrome alters the distribution of A-type lamins.

Authors:  Yuexia Wang; Cecilia Ostlund; Jason C Choi; Theresa C Swayne; Gregg G Gundersen; Howard J Worman
Journal:  Nucleus       Date:  2012-08-16       Impact factor: 4.197

5.  Skeletal and cardiac myopathies in mice lacking utrophin and dystrophin: a model for Duchenne muscular dystrophy.

Authors:  R M Grady; H Teng; M C Nichol; J C Cunningham; R S Wilkinson; J R Sanes
Journal:  Cell       Date:  1997-08-22       Impact factor: 41.582

6.  Dependence of diffusional mobility of integral inner nuclear membrane proteins on A-type lamins.

Authors:  Cecilia Ostlund; Teresa Sullivan; Colin L Stewart; Howard J Worman
Journal:  Biochemistry       Date:  2006-02-07       Impact factor: 3.162

7.  Depletion of extracellular signal-regulated kinase 1 in mice with cardiomyopathy caused by lamin A/C gene mutation partially prevents pathology before isoenzyme activation.

Authors:  Wei Wu; Shinichi Iwata; Shunichi Homma; Howard J Worman; Antoine Muchir
Journal:  Hum Mol Genet       Date:  2013-08-09       Impact factor: 6.150

Review 8.  Emerinopathy and laminopathy clinical, pathological and molecular features of muscular dystrophy with nuclear envelopathy in Japan.

Authors:  M N Astejada; K Goto; A Nagano; S Ura; S Noguchi; I Nonaka; I Nishino; Y K Hayashi
Journal:  Acta Myol       Date:  2007-12

9.  Lamin A/C haploinsufficiency causes dilated cardiomyopathy and apoptosis-triggered cardiac conduction system disease.

Authors:  Cordula M Wolf; Libin Wang; Ronny Alcalai; Anne Pizard; Patrick G Burgon; Ferhaan Ahmad; Megan Sherwood; Dorothy M Branco; Hiroko Wakimoto; Glenn I Fishman; Vincent See; Colin L Stewart; David A Conner; Charles I Berul; Christine E Seidman; J G Seidman
Journal:  J Mol Cell Cardiol       Date:  2007-12-03       Impact factor: 5.000

10.  Diet-induced obesity causes long QT and reduces transcription of voltage-gated potassium channels.

Authors:  Haiyan Huang; Vaibhav Amin; Michael Gurin; Elaine Wan; Edward Thorp; Shunichi Homma; John P Morrow
Journal:  J Mol Cell Cardiol       Date:  2013-03-19       Impact factor: 5.000

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  2 in total

1.  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

2.  Abolishing the prelamin A ZMPSTE24 cleavage site leads to progeroid phenotypes with near-normal longevity in mice.

Authors:  Yuexia Wang; Khurts Shilagardi; Trunee Hsu; Kamsi O Odinammadu; Takamitsu Maruyama; Wei Wu; Chyuan-Sheng Lin; Christopher B Damoci; Eric D Spear; Ji-Yeon Shin; Wei Hsu; Susan Michaelis; Howard J Worman
Journal:  Proc Natl Acad Sci U S A       Date:  2022-03-01       Impact factor: 12.779

  2 in total

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