Chen Chun-Rong, Hubbard Paul A, Salazar Larry M, McLachlan Sandra M, Murali Ramachandran, Rapoport Basil
Thyroid Autoimmune Disease Unit (C-R.C., L.M.S., S.M.M., B.R.) and Department of Biomedical Sciences (P.H., R.M.), Cedars-Sinai Research Institute and UCLA School of Medicine, Los Angeles, California 90048.
Mol Endocrinol. 2015 Jan;29(1):99-107. doi: 10.1210/me.2014-1257.
The TSH receptor (TSHR) A-subunit is more effective than the holoreceptor in inducing thyroid-stimulating antibodies (TSAb) that cause Graves' disease. A puzzling phenomenon is that 2 recombinant, eukaryotic forms of A-subunits (residues 22-289), termed active and inactive, are recognized mutually exclusively by pathogenic TSAb and mouse monoclonal antibody 3BD10, respectively. Understanding the structural difference between these TSHR A-subunit forms could provide insight into Graves' disease pathogenesis. The 3-dimensional structure of the active A-subunit (in complex with a human TSAb Fab, M22) is known, but the structural difference with inactive A-subunits is unknown. We solved the 3BD10 Fab 3-dimensional crystal structure. Guided by prior knowledge of a portion of its epitope, 3BD10 docked in silico with the known active TSHR-289 monomeric structure. Because both TSAb and 3BD10 recognize the active TSHR A-subunit monomer, this form of the molecule can be excluded as the basis for the active-inactive dichotomy, suggesting, instead a role for A-subunit quaternary structure. Indeed, in silico analysis revealed that M22, but not 3BD10, bound to a TSHR-289 trimer. In contrast, 3BD10, but not M22, bound to a TSHR-289 dimer. The validity of these models is supported experimentally by the temperature-dependent balance between active and inactive TSHR-289. In summary, we provide evidence for a structural basis to explain the conformational heterogeneity of TSHR A-subunits (TSHR-289). The pathophysiologic importance of these findings is that affinity maturation of pathogenic TSAb in Graves' disease is likely to involve a trimer of the shed TSHR A-subunit.
促甲状腺激素受体(TSHR)A亚基在诱导导致格雷夫斯病的促甲状腺素刺激抗体(TSAb)方面比全受体更有效。一个令人困惑的现象是,两种重组的真核形式的A亚基(第22 - 289位氨基酸残基),分别称为活性型和非活性型,分别被致病性TSAb和小鼠单克隆抗体3BD10相互排斥性地识别。了解这些TSHR A亚基形式之间的结构差异可能有助于深入了解格雷夫斯病的发病机制。活性A亚基(与人TSAb Fab,M22形成复合物)的三维结构是已知的,但与非活性A亚基的结构差异尚不清楚。我们解析了3BD10 Fab的三维晶体结构。根据其部分表位的先验知识,3BD10在计算机上与已知的活性TSHR - 289单体结构对接。由于TSAb和3BD10都识别活性TSHR A亚基单体,因此这种分子形式可以被排除作为活性 - 非活性二分法的基础,相反,提示A亚基四级结构起作用。实际上,计算机分析显示M22,但不是3BD10,与TSHR - 289三聚体结合。相反,3BD10,但不是M22,与TSHR - 289二聚体结合。活性和非活性TSHR - 289之间的温度依赖性平衡通过实验支持了这些模型的有效性。总之,我们提供了一个结构基础的证据来解释TSHR A亚基(TSHR - 289)的构象异质性。这些发现的病理生理重要性在于,格雷夫斯病中致病性TSAb的亲和力成熟可能涉及脱落的TSHR A亚基的三聚体。