Center for Molecular Medicine and Division of Endocrinology & Metabolism, University of Connecticut School of Medicine, Farmington, CT 06030-3101, USA.
Center for Molecular Medicine and Division of Endocrinology & Metabolism, University of Connecticut School of Medicine, Farmington, CT 06030-3101, USA.
Mol Cell Endocrinol. 2014 Apr 5;386(1-2):46-54. doi: 10.1016/j.mce.2013.09.005. Epub 2013 Sep 11.
Parathyroid neoplasia is most commonly due to benign parathyroid adenoma but rarely can be caused by malignant parathyroid carcinoma. Evidence suggests that parathyroid carcinomas rarely, if ever, evolve through an identifiable benign intermediate, with the notable exception of carcinomas associated with the familial hyperparathyroidism-jaw tumor syndrome. Several genes have been directly implicated in the pathogenesis of typical sporadic parathyroid adenoma; somatic mutations in the MEN1 tumor suppressor gene are the most frequent finding, and alterations in the cyclin D1/PRAD1 oncogene are also firmly established molecular drivers of sporadic adenomas. In addition, good evidence supports mutation in the CDKN1B/p27 cyclin-dependent kinase inhibitor (CDKI) gene, and in other CDKI genes as contributing to disease pathogenesis in this context. Somatic defects in additional genes, including β-catenin, POT1 and EZH2 may contribute to parathyroid adenoma formation but, for most, their ability to drive parathyroid tumorigenesis remains to be demonstrated experimentally. Further, genetic predisposition to sporadic presentations of parathyroid adenoma appears be conferred by rare, and probably low-penetrance, germline variants in CDKI genes and, perhaps, in other genes such as CASR and AIP. The HRPT2 tumor suppressor gene is commonly mutated in parathyroid carcinoma.
甲状旁腺肿瘤最常见的原因是良性甲状旁腺腺瘤,但很少由恶性甲状旁腺癌引起。有证据表明,甲状旁腺癌很少(如果有的话)通过可识别的良性中间阶段演变而来,家族性甲状旁腺功能亢进-颌骨肿瘤综合征相关的癌例外。一些基因已被直接牵连到典型的散发性甲状旁腺腺瘤的发病机制中;MEN1 肿瘤抑制基因的体细胞突变是最常见的发现,细胞周期蛋白 D1/PRAD1 癌基因的改变也是散发性腺瘤的明确分子驱动因素。此外,有充分的证据支持 CDKN1B/p27 细胞周期蛋白依赖性激酶抑制剂 (CDKI) 基因突变,以及其他 CDKI 基因在这种情况下导致疾病发病机制的突变。其他基因的体细胞缺陷,包括 β-连环蛋白、POT1 和 EZH2,可能有助于甲状旁腺腺瘤的形成,但对于大多数基因来说,它们驱动甲状旁腺肿瘤发生的能力仍有待实验证明。此外,散发性甲状旁腺腺瘤的遗传易感性似乎是由 CDKI 基因和其他基因(如 CASR 和 AIP)中的罕见(可能是低外显率)种系变体赋予的。HRPT2 肿瘤抑制基因在甲状旁腺癌中经常发生突变。