Lynch Henry T, Shaw Trudy G, Lynch Jane F
Department of Preventive Medicine, Creighton University School of Medicine, 2500 California Plaza, Omaha, NE 68178, USA.
Am J Med Genet C Semin Med Genet. 2004 Aug 15;129C(1):5-22. doi: 10.1002/ajmg.c.30026.
The hereditary predisposition to cancer dates historically to interest piqued by physicians as well as family members wherein striking phenotypic features were shown to cluster in families, inclusive of the rather grotesque cutaneous findings in von Recklinghausen's neurofibromatosis, which date back to the sixteenth century. The search for the role of primary genetic factors was heralded by studies at the infrahuman level, particularly on laboratory mouse strains with strong susceptibility to carcinogen-induced cancer, and conversely, with resistance to the same carcinogens. These studies, developed in the 19th and 20th centuries, continue today. This article traces the historical aspects of hereditary cancer dealing with identification and ultimate molecular genetic confirmation of commonly occurring cancers, particularly of the colon in the case of familial adenomatous polyposis and its attenuated form, both due to the APC germline mutation; the Lynch syndrome due to mutations in mismatch repair genes, the most common of which were found to be MSH2, MLH1, and MSH6 germline mutations; the hereditary breast-ovarian cancer syndrome with BRCA1 and BRCA2 germline mutations; the Li-Fraumeni (SBLA) syndrome due to the p53 mutation; and the familial atypical multiple mole melanoma in association with pancreatic cancer due to the CDKN2A (p16) germline mutation. These and other hereditary cancer syndromes have been discussed in some detail relevant to their characterization, which, for many conditions, took place in the late 18th century and, in the more modern molecular genetic era, during the past two decades. Emphasis has been placed upon the manner in which improved cancer control will emanate from these discoveries.
癌症的遗传易感性在历史上可追溯到医生以及家庭成员所引发的兴趣,其中显著的表型特征在家族中呈聚集性,包括可追溯到16世纪的冯·雷克林豪森神经纤维瘤病中相当怪异的皮肤表现。对主要遗传因素作用的探索始于对低等动物水平的研究,特别是对致癌物诱导癌症易感性强以及对相同致癌物具有抗性的实验小鼠品系的研究。这些在19世纪和20世纪开展的研究一直持续到今天。本文追溯了遗传性癌症的历史方面,涉及常见癌症的鉴定及最终的分子遗传学确认,特别是家族性腺瘤性息肉病及其弱化形式的结肠癌,二者均由APC种系突变引起;错配修复基因发生突变导致的林奇综合征,其中最常见的是MSH2、MLH1和MSH6种系突变;具有BRCA1和BRCA2种系突变的遗传性乳腺癌-卵巢癌综合征;由p53突变引起的李-弗劳梅尼(SBLA)综合征;以及由CDKN2A(p16)种系突变导致的家族性非典型多发性痣黑色素瘤与胰腺癌。这些以及其他遗传性癌症综合征已结合其特征进行了详细讨论,其中许多病症的特征描述在18世纪后期进行,而在更现代的分子遗传学时代,则是在过去二十年中进行的。重点放在了这些发现将如何带来癌症控制改善的方式上。