Baysal B E
Department of Psychiatry, The University of Pittsburgh Medical Center, 3811 O'Hara Street R1445, Pittsburgh, PA, 15213, USA.
J Med Genet. 2002 Sep;39(9):617-22. doi: 10.1136/jmg.39.9.617.
Paragangliomas are highly vascularised and often heritable tumours derived from paraganglia, a diffuse neuroendocrine system dispersed from skull base to the pelvic floor. The carotid body, a small oxygen sensing organ located at the bifurcation of the carotid artery in the head and neck and the adrenal medulla in the abdomen, are the most common tumour sites. It now appears that mutations in SDHB, SDHC, and SDHD, which encode subunits of mitochondrial complex II (succinate dehydrogenase; succinate-ubiquinone oxidoreductase), are responsible for the majority of familial paragangliomas and also for a significant fraction of non-familial tumours. Germline mutations in complex II genes are associated with the development of paragangliomas in diverse anatomical locations, including phaeochromocytomas, a finding that has important implications for the clinical management of patients and genetic counselling of families. Consequently, patients with a paraganglioma tumour, including phaeochromocytoma, and a complex II germline mutation should be diagnosed with hereditary paraganglioma, regardless of family history, anatomical location, or multiplicity of tumours. This short review attempts to bring together relevant genetic data on paragangliomas with a particular emphasis on head and neck paragangliomas and phaeochromocytomas.
副神经节瘤是高度血管化且通常具有遗传性的肿瘤,起源于副神经节,这是一个从颅底到盆底广泛分布的神经内分泌系统。颈动脉体,一个位于头颈部颈动脉分叉处的小型氧传感器官以及腹部的肾上腺髓质,是最常见的肿瘤发生部位。现在看来,编码线粒体复合物II(琥珀酸脱氢酶;琥珀酸-泛醌氧化还原酶)亚基的SDHB、SDHC和SDHD基因的突变,是大多数家族性副神经节瘤以及相当一部分非家族性肿瘤的病因。复合物II基因的种系突变与包括嗜铬细胞瘤在内的不同解剖部位的副神经节瘤的发生相关,这一发现对患者的临床管理和家庭遗传咨询具有重要意义。因此,患有副神经节瘤肿瘤(包括嗜铬细胞瘤)且存在复合物II种系突变的患者,无论家族史、解剖位置或肿瘤数量如何,均应诊断为遗传性副神经节瘤。本简短综述试图汇集有关副神经节瘤的相关遗传数据,特别强调头颈部副神经节瘤和嗜铬细胞瘤。