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本文引用的文献

1
Combined loss of LAP1B and LAP1C results in an early onset multisystemic nuclear envelopathy.LAP1B 和 LAP1C 联合缺失导致早发性多系统核纤层病。
Nat Commun. 2019 Feb 5;10(1):605. doi: 10.1038/s41467-019-08493-7.
2
The molecular architecture of lamins in somatic cells.体细胞中层粘连蛋白的分子结构。
Nature. 2017 Mar 9;543(7644):261-264. doi: 10.1038/nature21382. Epub 2017 Mar 1.
3
Nuclear envelope structural proteins facilitate nuclear shape changes accompanying embryonic differentiation and fidelity of gene expression.核膜结构蛋白促进伴随胚胎分化的核形态变化以及基因表达的保真度。
BMC Cell Biol. 2017 Jan 14;18(1):8. doi: 10.1186/s12860-017-0125-0.
4
Lamina-associated polypeptide 1 is dispensable for embryonic myogenesis but required for postnatal skeletal muscle growth.核纤层相关多肽1对胚胎期肌发生并非必需,但对出生后骨骼肌生长却是必需的。
Hum Mol Genet. 2017 Jan 1;26(1):65-78. doi: 10.1093/hmg/ddw368.
5
TOR1AIP1 as a cause of cardiac failure and recessive limb-girdle muscular dystrophy.TOR1AIP1作为心力衰竭和隐性肢带型肌营养不良的病因。
Neuromuscul Disord. 2016 Aug;26(8):500-3. doi: 10.1016/j.nmd.2016.05.013. Epub 2016 May 24.
6
Identification of a regulatory domain controlling the Nppa-Nppb gene cluster during heart development and stress.在心脏发育和应激过程中控制Nppa-Nppb基因簇的调控结构域的鉴定。
Development. 2016 Jun 15;143(12):2135-46. doi: 10.1242/dev.132019. Epub 2016 Apr 5.
7
Extracellular signal-regulated kinases 1/2 as regulators of cardiac hypertrophy.细胞外信号调节激酶1/2作为心脏肥大的调节因子。
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8
Tissue specific loss of A-type lamins in the gastrointestinal epithelium can enhance polyp size.胃肠道上皮中 A 型核纤层蛋白的组织特异性缺失可增大息肉尺寸。
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9
Severe dystonia, cerebellar atrophy, and cardiomyopathy likely caused by a missense mutation in TOR1AIP1.严重肌张力障碍、小脑萎缩和心肌病可能由TOR1AIP1基因的错义突变引起。
Orphanet J Rare Dis. 2014 Nov 26;9:174. doi: 10.1186/s13023-014-0174-9.
10
Depletion of lamina-associated polypeptide 1 from cardiomyocytes causes cardiac dysfunction in mice.心肌细胞中核纤层相关多肽1的缺失会导致小鼠心脏功能障碍。
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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.

DOI:10.1093/hmg/ddz082
PMID:31009944
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7590221/
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。