Mulligan L M, Eng C, Healey C S, Clayton D, Kwok J B, Gardner E, Ponder M A, Frilling A, Jackson C E, Lehnert H
Department of Pathology, University of Cambridge, UK.
Nat Genet. 1994 Jan;6(1):70-4. doi: 10.1038/ng0194-70.
We have analysed 118 families with inherited medullary thyroid carcinoma (MTC) for mutations of the RET proto-oncogene. These included cases of multiple endocrine neoplasia types 2A (MEN 2A) and 2B (MEN 2B) and familial MTC (FMTC). Mutations at one of 5 cysteines in the extracellular domain were found in 97% of patients with MEN 2A and 86% with FMTC but not in MEN 2B patients or normal controls. 84% of the MEN2A mutations affected codon 634. MEN 2A patients with a Cys634 to Arg substitution had a greater risk of developing parathyroid disease than those with other codon 634 mutations. Our data show a strong correlation between disease phenotype and the nature and position of the RET mutation, suggesting that a simple, constitutive activation of the RET tyrosine kinase is unlikely to explain the events leading to MEN 2A and FMTC.
我们分析了118个患有遗传性甲状腺髓样癌(MTC)的家族,以检测RET原癌基因的突变情况。这些家族包括2A型多发性内分泌腺瘤病(MEN 2A)、2B型多发性内分泌腺瘤病(MEN 2B)和家族性甲状腺髓样癌(FMTC)病例。细胞外结构域5个半胱氨酸之一发生突变的情况在97%的MEN 2A患者和86%的FMTC患者中被发现,但在MEN 2B患者或正常对照中未发现。84%的MEN2A突变影响密码子634。密码子634由半胱氨酸替换为精氨酸的MEN 2A患者发生甲状旁腺疾病的风险高于具有其他密码子634突变的患者。我们的数据显示疾病表型与RET突变的性质和位置之间存在很强的相关性,这表明RET酪氨酸激酶的简单组成性激活不太可能解释导致MEN 2A和FMTC的事件。