Little M, Wells C
Centre for Molecular and Cellular Biology, University of Queensland, St. Lucia, Australia.
Hum Mutat. 1997;9(3):209-25. doi: 10.1002/(SICI)1098-1004(1997)9:3<209::AID-HUMU2>3.0.CO;2-2.
Mutations in the WT1 gene were anticipated to explain the genetic basis of the childhood kidney cancer, Wilms tumour (WT). Six years on, we review 100 reports of intragenic WT1 mutations and examine the accompanying clinical phenotypes. While only 5% of sporadic Wilms' tumours have intragenic WT1 mutations, > 90% of patients with the Denys-Drash syndrome (renal nephropathy, gonadal anomaly, predisposition to WT) carry constitutional intragenic WT1 mutations. WT1 mutations have also been reported in juvenile granulosa cell tumour, non-asbestos related mesothelioma, desmoplastic small round cell tumour and, most recently, acute myeloid leukemia.
WT1基因的突变被认为可以解释儿童肾癌——威尔姆斯瘤(WT)的遗传基础。六年过去了,我们回顾了100篇关于WT1基因内突变的报告,并研究了与之相关的临床表型。虽然只有5%的散发性威尔姆斯瘤存在WT1基因内突变,但超过90%的患有迪尼-德拉斯综合征(肾病、性腺异常、易患WT)的患者携带先天性WT1基因内突变。WT1突变也在青少年颗粒细胞瘤、非石棉相关的间皮瘤、促结缔组织增生性小圆细胞肿瘤以及最近的急性髓系白血病中被报道。