Hengstschläger M, Rodman D M, Miloloza A, Hengstschläger-Ottnad E, Rosner M, Kubista M
Obstetrics and Gynecology, Prenatal Diagnosis and Therapy, University of Vienna, Währinger Gürtel 18-20, A-1090, Vienna, Austria.
Mutat Res. 2001 Jul;488(3):233-9. doi: 10.1016/s1383-5742(01)00058-8.
Two genes, TSC1 and TSC2, have been shown to be responsible for tuberous sclerosis (TSC). The detection of loss of heterozygosity of TSC1 or TSC2 in hamartomas, the growths characteristically occurring in TSC patients, suggested a tumor suppressor function for their gene products hamartin and tuberin. Studies analyzing ectopically modulated expression of TSC2 in human and rodent cells together with the finding that a homolog of TSC2 regulates the Drosophila cell cycle suggest that TSC is a disease of proliferation/cell cycle control. We discuss this question including very recent data obtained from analyzing mice expressing a modulated TSC2 transgene, and from studying the effects of deregulated TSC1 expression. Elucidation of the cellular functions of these proteins will form the basis of a better understanding of how mutations in these genes cause the disease and for the development of new therapeutic strategies.
已证明两个基因TSC1和TSC2与结节性硬化症(TSC)有关。在错构瘤(TSC患者特有的生长物)中检测到TSC1或TSC2杂合性缺失,提示其基因产物错构素和结节素具有肿瘤抑制功能。对人和啮齿动物细胞中TSC2异位调节表达的分析研究,以及TSC2的一个同源物调节果蝇细胞周期这一发现,提示TSC是一种增殖/细胞周期调控疾病。我们将讨论这个问题,包括分析表达调节型TSC2转基因的小鼠以及研究TSC1表达失调的影响所获得的最新数据。阐明这些蛋白质的细胞功能将为更好地理解这些基因中的突变如何导致该疾病以及开发新的治疗策略奠定基础。