Lynch Henry T, Lynch Jane F, Shaw Trudy G
Department of Preventive Medicine and Public Health, Creighton University, Omaha, NE.
Gastrointest Cancer Res. 2011 Jul;4(4 Suppl 1):S9-S17.
The rapid growth of molecular genetics and its attendant germline mutation discoveries has enabled identification of persons who are at an inordinately high cancer risk and, therefore, ideal candidates for prevention. However, one must fully appreciate the extensive genotypic and phenotypic heterogeneity that exists in hereditary cancer. Once the causative germline mutation has been identified in a patient, high-risk members of the family can be similarly tested and identified and provided highly targeted surveillance and management opportunities. DNA testing can change the individual's presumed risk status and affect decision making by patients and their physicians regarding surveillance and management. Our purpose is to describe familial/hereditary cancers of the gastrointestinal tract, including familial Barrett's esophagus, hereditary diffuse gastric cancer, gastrointestinal stromal tumors, familial adenomatous polyposis and desmoid tumors, Lynch syndrome, small bowel cancer, and familial pancreatic cancer. We use our discussion of Lynch syndrome as a model for diagnostic and clinical translation strategies for all hereditary gastrointestinal tract cancers, which clearly can then be extended to cancer of all anatomic sites. Highly pertinent questions from the patient's perspective include the following: What kind of counseling will be provided to a patient with a Lynch syndrome mutation, and should that counseling be mandatory? Does the proband have the responsibility to inform relatives about the familial mutation, even if the relatives do not want to know whether they carry it? Is the patient is responsible for notifying family members that a parent or sibling has Lynch syndrome? Can notification be forced and, if so, under what circumstances? These questions point out the need for criteria regarding which family members to inform and how to inform them.
分子遗传学的迅速发展及其相关的种系突变发现,使得识别出患癌风险极高的人群成为可能,因此他们也是预防的理想对象。然而,必须充分认识到遗传性癌症中存在的广泛的基因型和表型异质性。一旦在患者中确定了致病种系突变,就可以对家族中的高危成员进行类似的检测和识别,并为他们提供高度针对性的监测和管理机会。DNA检测可以改变个体假定的风险状态,并影响患者及其医生关于监测和管理的决策。我们的目的是描述胃肠道的家族性/遗传性癌症,包括家族性巴雷特食管、遗传性弥漫性胃癌、胃肠道间质瘤、家族性腺瘤性息肉病和硬纤维瘤、林奇综合征、小肠癌和家族性胰腺癌。我们以对林奇综合征的讨论作为所有遗传性胃肠道癌症诊断和临床转化策略的模型,显然这一策略随后可扩展到所有解剖部位的癌症。从患者角度来看,高度相关的问题包括以下几点:对于携带林奇综合征突变的患者将提供何种咨询,这种咨询是否应该是强制性的?先证者是否有责任告知亲属家族性突变情况,即使亲属不想知道自己是否携带该突变?患者是否有责任告知家庭成员其父母或兄弟姐妹患有林奇综合征?能否强制进行告知,如果可以,在什么情况下?这些问题指出了需要制定关于告知哪些家庭成员以及如何告知他们的标准。