Klein M A, Rüedi D, Nozaki M, Dell E W, Diserens A C, Seelentag W, Janzer R C, Aguzzi A, Hegi M E
Institute of Neuropathology, University Hospital of Zurich, Switzerland.
Oncogene. 2000 Nov 9;19(47):5329-37. doi: 10.1038/sj.onc.1203941.
p53-germline mutations located in the core DNA-binding domain have been associated with a more dominant tumor penetrance especially for breast cancer and brain tumors. We previously reported an unusual accumulation of CNS tumors associated with a unique p53 germline mutation, Y236delta (deletion of codon 236). To test whether this tissue-specific tumor predisposition reflects a gain-of-function activity of Y236delta, we generated transgenic mice expressing Y236delta in astrocytes using the regulatory elements of the glial fibrillary acidic protein (GFAP) gene. After transplacental exposure to N-ethyl-N-nitrosourea (25 mg/kg BW) brain tumors developed in 18% (7/39) of GFAP-Y236delta transgenic p53-/- mice, while in p53+/- mice the incidence was 28% (11/40) (P>0.3). However, the mean tumor latency for GFAP-Y236delta/p53+/- mice was significantly shorter than for p53+/- mice, with 19.9 weeks vs 31.6 weeks (P=0.039), respectively. Taken together, cell specific expression of Y236delta results in an acceleration of tumor progression but does not confer a higher tumor penetrance. Conceivably, the transdominant effect of Y236delta provided a growth advantage early in the progression of neoplastic cells, since the endogenous p53 wild-type allele was lost in all brain tumors independent of the genotype. This reflects well observations from human astrocytic neoplasms with p53 mutations.
位于核心DNA结合结构域的p53种系突变与更强的肿瘤显性相关,尤其是对于乳腺癌和脑肿瘤。我们之前报道过一种与独特的p53种系突变Y236delta(密码子236缺失)相关的中枢神经系统肿瘤异常聚集现象。为了测试这种组织特异性肿瘤易感性是否反映了Y236delta的功能获得活性,我们利用胶质纤维酸性蛋白(GFAP)基因的调控元件,生成了在星形胶质细胞中表达Y236delta的转基因小鼠。经胎盘暴露于N-乙基-N-亚硝基脲(25mg/kg体重)后,18%(7/39)的GFAP-Y236delta转基因p53-/-小鼠发生了脑肿瘤,而在p53+/-小鼠中,发病率为28%(11/40)(P>0.3)。然而,GFAP-Y236delta/p53+/-小鼠的平均肿瘤潜伏期明显短于p53+/-小鼠,分别为19.9周和31.6周(P=0.039)。综上所述,Y236delta的细胞特异性表达导致肿瘤进展加速,但并未赋予更高的肿瘤显性。可以想象,Y236delta的反式显性效应在肿瘤细胞进展早期提供了生长优势,因为在所有脑肿瘤中,内源性p53野生型等位基因均缺失,与基因型无关。这很好地反映了对具有p53突变的人类星形细胞瘤的观察结果。