Center for Interdisciplinary Sciences, Tata Institute of Fundamental Research, Gopanpally, Hyderabad 500107, India.
Center for Interdisciplinary Sciences, Tata Institute of Fundamental Research, Gopanpally, Hyderabad 500107, India; Department of Chemical Sciences, Tata Institute of Fundamental Research, 1, Homi Bhabha Road, Colaba, Mumbai 400005, India; Indian Institute of Science Education and Research, Berhampur, Odisha 760010, India.
J Struct Biol. 2019 Mar 1;205(3):72-78. doi: 10.1016/j.jsb.2019.02.003. Epub 2019 Feb 12.
A recently identified mutant of human γS-crystallin, G57W is associated with dominant congenital cataracts, the familial determinate of childhood blindness worldwide. To investigate the structural and functional changes that mediate the effect of this cataract-related mutant to compromise eye lens transparency and cause lens opacification in children, we recently reported complete sequence-specific resonance assignments of γS-G57W using a suite of heteronuclear NMR experiments. As a follow up, we have determined the 3D structure of γS-G57W and studied its conformational dynamics by solution NMR spectroscopy. Our structural dynamics results reveal greater flexibility of the N-terminal domain, which undergoes site-specific structural changes to accommodate W57, than its C-terminal counterpart. Our structural inferences that the unusual solvent exposure of W57 is associated with rearrangement of the N-terminal domain suggest an efficient pathway for increased aggregation in γS-G57W and illuminates the molecular dynamics underlying cataractogenic aggregation of lens crystallins in particular and aggregation of proteins in general.
最近发现的一种人 γS-晶体蛋白的突变体 G57W 与显性先天性白内障有关,这种白内障是全世界儿童致盲的家族决定因素。为了研究导致这种与白内障相关的突变体影响眼睛晶状体透明度并导致儿童晶状体混浊的结构和功能变化,我们最近使用一系列异核 NMR 实验报告了 γS-G57W 的完整序列特异性共振分配。作为后续研究,我们确定了 γS-G57W 的 3D 结构,并通过溶液 NMR 光谱研究了其构象动力学。我们的结构动力学结果表明,N 端结构域的柔韧性更大,其经历了特定于位置的结构变化以适应 W57,而 C 端结构域则没有。我们的结构推断表明,W57 的异常溶剂暴露与 N 端结构域的重排有关,这表明在 γS-G57W 中增加聚集的有效途径,并阐明了晶状体晶体特别是蛋白质聚集的分子动力学基础。