You Min, Li Edwin, Hristova Kalina
Department of Materials Science and Engineering, Johns Hopkins University, Baltimore, Maryland 21218, USA.
Biochemistry. 2006 May 2;45(17):5551-6. doi: 10.1021/bi060113g.
The Gly380 --> Arg mutation in the TM domain of fibroblast growth factor receptor 3 (FGFR3) of the RTK family is linked to achondroplasia, the most common form of human dwarfism. The molecular mechanism of pathology induction is under debate, and two different mechanisms have been proposed to contribute to pathogenesis: (1) Arg380-mediated FGFR3 dimer stabilization and (2) slow downregulation of the activated mutant receptors. Here we show that the Gly380 --> Arg mutation does not alter the dimerization energetics of the FGFR3 transmembrane domain in detergent micelles or in lipid bilayers. This result indicates that pathogenesis in achondroplasia cannot be explained simply by a higher dimerization propensity of the mutant FGFR3 TM domain, thus highlighting the importance of the observed slow downregulation in phenotype induction.
受体酪氨酸激酶(RTK)家族成纤维细胞生长因子受体3(FGFR3)跨膜结构域中的Gly380→Arg突变与软骨发育不全有关,软骨发育不全是人类侏儒症最常见的形式。病理诱导的分子机制仍在争论中,并且已经提出了两种不同的机制来促成发病机制:(1)Arg380介导的FGFR3二聚体稳定化和(2)活化的突变受体的下调减缓。在这里,我们表明Gly380→Arg突变不会改变去污剂胶束或脂质双层中FGFR3跨膜结构域的二聚化能量。该结果表明,软骨发育不全的发病机制不能简单地通过突变的FGFR3跨膜结构域更高的二聚化倾向来解释,从而突出了在表型诱导中观察到的下调减缓的重要性。