Han Xue, Mihailescu Mihaela, Hristova Kalina
Department of Materials Science and Engineering, Johns Hopkins University, Baltimore, Maryland, USA.
Biophys J. 2006 Nov 15;91(10):3736-47. doi: 10.1529/biophysj.106.092247. Epub 2006 Sep 1.
Achondroplasia, the most common form of human dwarfism, is due to a G380R mutation in the transmembrane domain of fibroblast growth factor receptor 3 (FGFR3) in >97% of the studied cases. While the molecular mechanism of pathology induction is under debate, the structural consequences of the mutation have not been studied. Here we use neutron diffraction to determine the disposition of FGFR3 transmembrane domain in fluid lipid bilayers, and investigate whether the G380R mutation affects the topology of the protein in the bilayer. Our results demonstrate that, in a model system, the G380R mutation induces a shift in the segment that is embedded in the membrane. The center of the hydrocarbon core-embedded segment in the mutant is close to the midpoint between R380 and R397, supporting previous measurements of arginine insertion energetics into the endoplasmic reticulum. The presented results further our knowledge about basic amino-acid insertion into bilayers, and may lead to new insights into the mechanism of pathogenesis in achondroplasia.
软骨发育不全是人类侏儒症最常见的形式,在97%以上的研究病例中,是由成纤维细胞生长因子受体3(FGFR3)跨膜结构域中的G380R突变引起的。虽然病理诱导的分子机制仍在争论中,但该突变的结构后果尚未得到研究。在这里,我们使用中子衍射来确定FGFR3跨膜结构域在流体脂质双层中的排列,并研究G380R突变是否会影响该蛋白在双层中的拓扑结构。我们的结果表明,在一个模型系统中,G380R突变会导致嵌入膜中的片段发生移位。突变体中嵌入烃核的片段中心靠近R380和R397之间的中点,这支持了先前关于精氨酸插入内质网能量学的测量。所呈现的结果增进了我们对碱性氨基酸插入双层的认识,并可能为软骨发育不全的发病机制带来新的见解。