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Progerin in muscle leads to thermogenic and metabolic defects via impaired calcium homeostasis

  • Wan Ping Wang
  • , Jing Ya Wang
  • , Wen Hsin Lin
  • , Cheng Heng Kao
  • , Ming Chun Hung
  • , Yuan Chi Teng
  • , Ting Fen Tsai
  • , Ya Hui Chi*
  • *此作品的通信作者
  • National Health Research Institutes Taiwan
  • National Yang Ming Chiao Tung University
  • China Medical University Taichung

研究成果: 期刊稿件 › 文章 › 同行評審

22 引文 斯高帕斯(Scopus)

摘要

Mutations in lamin A (LMNA) are responsible for a variety of human dystrophic and metabolic diseases. Here, we created a mouse model in which progerin, the lamin A mutant protein that causes Hutchinson–Gilford progeria syndrome (HGPS), can be inducibly overexpressed. Muscle-specific overexpression of progerin was sufficient to induce muscular dystrophy and alter whole-body energy expenditure, leading to premature death. Intriguingly, sarcolipin (Sln), an endoplasmic reticulum (ER)-associated protein involved in heat production, is upregulated in progerin-expressing and Lmna knockout (Lmna−/−) skeletal muscle. The depletion of Sln accelerated the early death of Lmna−/− mice. An examination at the molecular level revealed that progerin recruits Sln and Calnexin to the nuclear periphery. Furthermore, progerin-expressing myoblasts presented enhanced store-operated Ca2+ entry, as well as increased co-localization of STIM1 and ORAI1. These findings suggest that progerin dysregulates calcium homeostasis through an interaction with a subset of ER-associated proteins, resulting in thermogenic and metabolic abnormalities.

原文英語
文章編號e13090
期刊Aging Cell
卷19
發行號2
DOIs
出版狀態已出版 - 01 02 2020

文獻附註

Publisher Copyright:
© 2019 The Authors. Aging Cell published by the Anatomical Society and John Wiley & Sons Ltd.

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