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lamin A/C、emerin 和核膜相关多肽 1 联合基因缺失的小鼠的产后发育。

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

机构信息

Department of Medicine and.

Department of Pathology and Cell Biology, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY, USA.

出版信息

Hum Mol Genet. 2019 Aug 1;28(15):2486-2500. doi: 10.1093/hmg/ddz082.

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.

摘要

编码核纤层蛋白 A/C 的 LMNA 和编码弹力蛋白的 EMD 的突变会导致心肌病和肌肉萎缩症。 Lmna 基因敲除小鼠会出现这些疾病,且其寿命为 7-8 周。 Emd 基因敲除小鼠没有明显的病理表现,且具有正常的骨骼肌,但存在再生缺陷。我们生成了 Lmna 和 Emd 均缺失的生殖系敲除小鼠,以确定这两种编码蛋白缺失的综合影响。没有核纤层蛋白 A/C 和弹力蛋白的小鼠按预期的孟德尔比例出生,出生时大体正常,但寿命比仅缺失核纤层蛋白 A/C 的小鼠短。然而,这些同时缺乏核纤层蛋白 A/C 和弹力蛋白的小鼠在左心室功能、心脏超微结构或心电图参数方面没有明显差异,除了缺乏核纤层蛋白 A/C 和弹力蛋白的小鼠心率较慢。这两种小鼠的骨骼肌也受到类似的影响。同时缺乏弹力蛋白的 Lmna+/- 小鼠也至少能活到 1 岁,与 Lmna+/- 小鼠相比,它们的生长、心脏或骨骼肌没有显著差异。编码核膜相关蛋白 1 的小鼠基因缺失会导致产前死亡;然而,杂合缺失该基因且同时缺乏核纤层蛋白 A/C 和弹力蛋白的小鼠按预期的孟德尔比例出生,但寿命比仅缺乏核纤层蛋白 A/C 和弹力蛋白的小鼠短。这些结果表明,同时缺乏三种相互作用的核膜蛋白的小鼠具有正常的胚胎发育,而早期的出生后缺陷主要是由核纤层蛋白 A/C 或核膜相关多肽 1 的缺失而非弹力蛋白的缺失驱动的。

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The molecular architecture of lamins in somatic cells.体细胞中层粘连蛋白的分子结构。
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