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核纤层蛋白 A 骨骼肌萎缩症突变改变 Ig 折叠结构域的结构和动力学。

Skeletal Muscle Dystrophy mutant of lamin A alters the structure and dynamics of the Ig fold domain.

机构信息

Biophysics & Structural Genomics Division, Saha Institute of Nuclear Physics, Homi Bhabha National Institute, 1/AF Bidhan nagar, Kolkata, 700064, West Bengal, India.

Department of Biochemistry, University of Calcutta, 35 Ballygunge Circular Road, Kolkata, 700019, West Bengal, India.

出版信息

Sci Rep. 2018 Sep 14;8(1):13793. doi: 10.1038/s41598-018-32227-2.

Abstract

Mutations in the different domains of A-type lamin proteins cause a diverse plethora of diseases collectively termed as laminopathies which can affect multiple organs. Ig fold is one such domain of lamin A which is implicated in numerous nuclear interactions wherein the mutations lead to different laminopathies. W514R is one such mutation in the Ig fold which leads to severe phenotypes in Skeletal Muscle Dystrophy (SMD) which is a class of laminopathies. In this report, we elucidated gross alterations in structure and dynamics at the level of individual amino acids. These studies indicate altered conformational features of residues in the close vicinity of W514. Imaging of mammalian cells transfected with the mutant have shown distinct perturbation of the nuclear meshwork with concomitant alteration in nuclear interactions as a result of increased oligomerization of Ig W514R. Hence, this novel approach of amalgamating theoretical and experimental procedures to predict the severity of a mutant in the context of laminopathies could be extended for numerous lamin A mutants.

摘要

A 型层粘连蛋白蛋白不同结构域的突变导致多种疾病的统称,称为层粘连蛋白病,可影响多个器官。Ig 折叠是层粘连蛋白 A 的一个结构域,它参与了许多核相互作用,其中的突变导致了不同的层粘连蛋白病。Ig 折叠中的 W514R 突变导致严重的骨骼肌肉营养不良(SMD)表型,这是一种层粘连蛋白病。在本报告中,我们阐明了在单个氨基酸水平上结构和动力学的明显变化。这些研究表明,W514 附近残基的构象特征发生了改变。转染突变体的哺乳动物细胞的成像显示,核网格的明显扰动,以及核相互作用的改变,这是由于 Ig W514R 的寡聚化增加所致。因此,这种将理论和实验程序相结合的新方法来预测层粘连蛋白病中突变体的严重程度,可以扩展到许多层粘连蛋白 A 突变体。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/584d/6138676/6d87b1abdb60/41598_2018_32227_Fig1_HTML.jpg

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