Li F P, Breslow N E, Morgan J M, Ghahremani M, Miller G A, Grundy P E, Green D M, Diller L R, Pelletier J
Dana-Farber Cancer Institute, Boston, Massachusetts 02115, USA.
Med Pediatr Oncol. 1996 Nov;27(5):404-7. doi: 10.1002/(SICI)1096-911X(199611)27:5<404::AID-MPO3>3.0.CO;2-Q.
We conducted a comparative study of the prevalence of germline WT1 mutations in patients with Wilms' tumor. Patients in Group 1 have familial Wilms' tumor, bilateral disease, associated urogenital anomalies, and/or second cancers. Those in Group 2 are unilateral, sporadic Wilms' patients without other associated conditions. Patients with aniridia or Denys-Drash syndrome are known to have WT1 alterations, and are excluded from this study. Preliminary results on 96 subjects show that the overall germline WT1 mutation frequency is low (< 5%). The work to date establishes the feasibility of identifying patients with germline WT1 mutations and, in the future, offering genetic predisposition testing to at-risk relatives. However, genetic predisposition testing of children for WT1 mutations raises many ethical, legal, and psychosocial issues; research is needed to evaluate risks and benefits.
我们对肾母细胞瘤患者种系WT1突变的患病率进行了一项比较研究。第1组患者患有家族性肾母细胞瘤、双侧疾病、相关泌尿生殖系统异常和/或第二癌症。第2组患者为无其他相关病症的单侧、散发性肾母细胞瘤患者。已知患有无虹膜或Denys-Drash综合征的患者有WT1改变,因此被排除在本研究之外。对96名受试者的初步结果表明,种系WT1突变的总体频率较低(<5%)。迄今为止的工作确定了识别种系WT1突变患者以及未来为有风险的亲属提供遗传易感性检测的可行性。然而,对儿童进行WT1突变的遗传易感性检测引发了许多伦理、法律和社会心理问题;需要开展研究以评估风险和益处。