Habib Samy L, Al-Obaidi Noor Y, Nowacki Maciej, Pietkun Katarzyna, Zegarska Barbara, Kloskowski Tomasz, Zegarski Wojciech, Drewa Tomasz, Medina Edward A, Zhao Zhenze, Liang Sitai
Geriatric Research Education and Clinical Center, South Texas, Veterans Healthcare System; Department of Cellular and Structural Biology, University of Texas Health Science Center, San Antonio, Texas 78229, USA.
Department of Cellular and Structural Biology, University of Texas Health Science Center, San Antonio, Texas 78229, USA.
J Cancer. 2016 Jul 21;7(12):1621-1631. doi: 10.7150/jca.14747. eCollection 2016.
Tuberous sclerosis complex (TSC) is an autosomal dominant and multi-system genetic disorder in humans. TSC affects around 25,000 to 40,000 individuals in the United States and about 1 to 2 million individuals worldwide, with an estimated prevalence of one in 6,000 newborns. TSC occurs in all races and ethnic groups, and in both genders. TSC is caused by defects or mutations in two genes, TSC1 and TSC2. Loss of TSC1/TSC2 leads to dysregulation of mTOR, resulting in aberrant cell differentiation and development, and abnormal enlargement of cells. TSC is characterized by the development of benign and/or malignant tumors in several organs including renal/liver angiomyolipomas, facial angiofibroma, lymphangiomyomatosis, cardiac rhabdomyomas, retinal astrocytic, renal cell carcinoma, and brain subependymal giant cell astrocytomas (SEGA). In addition, TSC disease causes disabling neurologic disorders, including epilepsy, mental retardation and autism. Particularly problematic are the development of renal angiomyolipomas, which tend to be larger, bilateral, multifocal and present at a younger age compared with sporadic forms. In addition, SEGA block the flow of fluid within the brain, causing a buildup of fluid and pressure that leads to blurred vision and seizures. In the current review, we describe the pathology of TSC disease in key organs and summarize the use of mTOR inhibitors to treat tumors in TSC patients.
结节性硬化症(TSC)是一种人类常染色体显性多系统遗传病。在美国,约有25000至40000人受TSC影响,全球约有100万至200万人受其影响,估计新生儿患病率为1/6000。TSC在所有种族和民族以及男女中均有发生。TSC由TSC1和TSC2这两个基因的缺陷或突变引起。TSC1/TSC2缺失会导致mTOR失调,从而导致细胞分化和发育异常以及细胞异常增大。TSC的特征是在多个器官中出现良性和/或恶性肿瘤,包括肾/肝血管平滑肌脂肪瘤、面部血管纤维瘤、淋巴管平滑肌瘤病、心脏横纹肌瘤、视网膜星形细胞瘤、肾细胞癌和脑室管膜下巨细胞星形细胞瘤(SEGA)。此外,TSC疾病会导致致残性神经障碍,包括癫痫、智力迟钝和自闭症。特别成问题的是肾血管平滑肌脂肪瘤的发展,与散发性形式相比,其往往更大、双侧、多灶且发病年龄更小。此外,SEGA会阻碍脑内液体流动,导致液体和压力积聚,进而导致视力模糊和癫痫发作。在本综述中,我们描述了TSC疾病在关键器官中的病理,并总结了mTOR抑制剂在治疗TSC患者肿瘤中的应用。