Vanharanta Sakari, Buchta Mary, McWhinney Sarah R, Virta Sanna K, Peçzkowska Mariola, Morrison Carl D, Lehtonen Rainer, Januszewicz Andrzej, Järvinen Heikki, Juhola Matti, Mecklin Jukka-Pekka, Pukkala Eero, Herva Riitta, Kiuru Maija, Nupponen Nina N, Aaltonen Lauri A, Neumann Hartmut P H, Eng Charis
Department of Medical Genetics, Biomedicum Helsinki, University of Helsinki, Finland.
Am J Hum Genet. 2004 Jan;74(1):153-9. doi: 10.1086/381054. Epub 2003 Dec 18.
Hereditary paraganglioma syndrome has recently been shown to be caused by germline heterozygous mutations in three (SDHB, SDHC, and SDHD) of the four genes that encode mitochondrial succinate dehydrogenase. Extraparaganglial component neoplasias have never been previously documented. In a population-based registry of symptomatic presentations of phaeochromocytoma/paraganglioma comprising 352 registrants, among whom 16 unrelated registrants were SDHB mutation positive, one family with germline SDHB mutation c.847-50delTCTC had two members with renal cell carcinoma (RCC), of solid histology, at ages 24 and 26 years. Both also had paraganglioma. A registry of early-onset RCCs revealed a family comprising a son with clear-cell RCC and his mother with a cardiac tumor, both with the germline SDHB R27X mutation. The cardiac tumor proved to be a paraganglioma. All RCCs showed loss of the remaining wild-type allele. Our observations suggest that germline SDHB mutations can predispose to early-onset kidney cancers in addition to paragangliomas and carry implications for medical surveillance.
遗传性副神经节瘤综合征最近被证明是由编码线粒体琥珀酸脱氢酶的四个基因中的三个(SDHB、SDHC和SDHD)的种系杂合突变引起的。此前从未有过神经节外成分肿瘤的记录。在一个基于人群的嗜铬细胞瘤/副神经节瘤症状表现登记处,共有352名登记者,其中16名无亲缘关系的登记者SDHB突变呈阳性,一个种系SDHB突变c.847-50delTCTC的家族中有两名成员,分别在24岁和26岁时患实性组织学类型的肾细胞癌(RCC)。两人也都患有副神经节瘤。一项早发性肾细胞癌登记显示,一个家族中,儿子患透明细胞肾细胞癌,母亲患心脏肿瘤,两人都有SDHB R27X种系突变。心脏肿瘤经证实为副神经节瘤。所有肾细胞癌均显示剩余野生型等位基因缺失。我们的观察结果表明,种系SDHB突变除了易患副神经节瘤外,还可能导致早发性肾癌,这对医学监测具有重要意义。