Hengstschläger Markus, Rosner Margit, Fountoulakis Michael, Lubec Gert
Medical Genetics, Department of Obstetrics and Gynecology, Medical University of Vienna, A-1090 Vienna, Austria.
Int J Oncol. 2005 Sep;27(3):831-8.
The genes, TSC1 on chromosome 9q34, encoding hamartin, and TSC2 on chromosome 16p13.3, encoding tuberin, are responsible for tuberous sclerosis (TSC). TSC is an autosomal dominant tumor suppressor gene syndrome affecting about 1 in 6000 individuals. It is characterized by mental retardation and epilepsy. A variety of tumors characteristically occur in different organs of TSC patients and are believed to result from defects in cell cycle/cell size control. Hamartin and tuberin form a complex providing a tentative explanation for the similar disease phenotype in TSC patients with mutations in either of these genes. Beside overlap in many features of patients with TSC1 and TSC2 mutations, data accumulated providing evidence for specific clinical differences. In this study, we performed a proteomic approach of two-dimensional gel electrophoresis with subsequent mass spectrometrical identification of protein spots after ectopic overexpression of human TSC1 or TSC2. We found the protein levels of the calumenin precursor; the complement component 1; heterogeneous nuclear ribonucleoproteins, C1/C2; heterogeneous nuclear ribonucleoprotein, C1-like protein; nascent polypeptide-associated complex-alpha; proteasome subunit alpha type 5; reticulocalbin 1 precursor; translationally-controlled tumor protein; UV excision repair protein, RAD23 homolog B; elongation factor 1-delta; and the eukaryotic initiation factors, eIF-4A-like NUK-34 and eIF-6; to be deregulated upon ectopic TSC gene expression. These findings suggest that deregulation of the control of these new target proteins might contribute to the development of tubers/hamartomas in tuberous sclerosis patients. The data are presented and discussed in the context of the published literature on proteomic approaches for the identification of targets of the TSC genes.
位于9号染色体长臂3区4带的TSC1基因编码错构瘤蛋白,位于16号染色体短臂1区3带3亚带的TSC2基因编码结节蛋白,这两个基因与结节性硬化症(TSC)有关。TSC是一种常染色体显性肿瘤抑制基因综合征,每6000人中约有1人受其影响。其特征为智力迟钝和癫痫。TSC患者的不同器官中会出现各种典型肿瘤,据信这是细胞周期/细胞大小控制缺陷所致。错构瘤蛋白和结节蛋白形成复合物,这为TSC患者中这两个基因任一发生突变时出现相似疾病表型提供了一种初步解释。除了TSC1和TSC2突变患者在许多特征上存在重叠外,积累的数据也为特定临床差异提供了证据。在本研究中,我们采用二维凝胶电泳的蛋白质组学方法,在异位过表达人TSC1或TSC2后,通过质谱鉴定蛋白质斑点。我们发现,在异位表达TSC基因后,钙网蛋白前体、补体成分1、不均一核核糖核蛋白C1/C2、不均一核核糖核蛋白C1样蛋白、新生多肽相关复合物α、蛋白酶体亚基α5型、网质钙结合蛋白1前体、翻译控制肿瘤蛋白、紫外线切除修复蛋白RAD23同源物B、延伸因子1-δ以及真核起始因子eIF-4A样NUK-34和eIF-6的蛋白质水平发生了失调。这些发现表明,这些新靶蛋白的调控失调可能促成结节性硬化症患者中结节/错构瘤的形成。我们结合已发表的关于蛋白质组学方法鉴定TSC基因靶点的文献,展示并讨论了这些数据。