Dannenberg Hilde, Komminoth Paul, Dinjens Winand N M, Speel Ernst Jan M, de Krijger Ronald R
Department of Pathology, Josephine Nefkens Institute, Erasmus MC Rotterdam, The Netherlands.
Endocr Pathol. 2003 Winter;14(4):329-50. doi: 10.1385/ep:14:4:329.
Pheochromocytomas and paragangliomas are neuroendocrine neoplasias of neural crest origin. Genetic mutations that are characterized in other human neoplasms are rarely seen in these tumors. About 10% of the patients with pheochromocytomas and paragangliomas present with a family history of von Hippel-Lindau disease (VHL), Multiple endocrine neoplasia type 2 (MEN2), one of the three familial paraganglioma syndromes (PGL; PGL1, PGL3, PGL4), or neurofibromatosis type 1 (NF1). In an even higher percentage, a genetic predisposition is involved in the development of these tumors. The genes of hereditary tumor syndromes such as the aforementioned ones are also ideal to study the molecular pathogenesis in the sporadic counterparts. Many studies have been undertaken to identify important secondary genetic events that contribute to the tumorigenesis of pheochromocytoma or paraganglioma, but a comprehensive review of these data is lacking. Recent findings of CGH and LOH studies provided new starting points to unravel the pathogenesis and progression of these tumors. This review presents an overview of our current understanding of the molecular pathogenesis of pheochromocytoma and paraganglioma.
嗜铬细胞瘤和副神经节瘤是起源于神经嵴的神经内分泌肿瘤。在其他人类肿瘤中所具有特征的基因突变在这些肿瘤中很少见。约10%的嗜铬细胞瘤和副神经节瘤患者有冯·希佩尔-林道病(VHL)、2型多发性内分泌肿瘤(MEN2)、三种家族性副神经节瘤综合征(PGL;PGL1、PGL3、PGL4)之一或1型神经纤维瘤病(NF1)的家族病史。在更高比例的患者中,遗传易感性参与了这些肿瘤的发生发展。诸如上述的遗传性肿瘤综合征相关基因也是研究散发性肿瘤分子发病机制的理想对象。已经开展了许多研究来确定导致嗜铬细胞瘤或副神经节瘤肿瘤发生的重要继发性遗传事件,但缺乏对这些数据的全面综述。比较基因组杂交(CGH)和杂合性缺失(LOH)研究的最新发现为阐明这些肿瘤的发病机制和进展提供了新的起点。本综述概述了我们目前对嗜铬细胞瘤和副神经节瘤分子发病机制的理解。