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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