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可能预防结节性硬化症病变。

Possible prevention of tuberous sclerosis complex lesions.

机构信息

Department of Science, The Children's Memorial Health Institute, Warsaw, Poland.

出版信息

Pediatrics. 2013 Jul;132(1):e239-42. doi: 10.1542/peds.2012-3607. Epub 2013 Jun 3.

Abstract

Tuberous sclerosis complex (TSC) is a genetic disorder characterized by mammalian target of rapamycin (mTOR) activation and growth of benign tumors. Some TSC lesions, such as cardiac rhabdomyomas and cortical tubers in the brain, occur in fetuses, and some, such as renal angiomyolipomas (AMLs) and skin angiofibromas, develop over years. Recently, the mTOR inhibitor everolimus was shown to be effective in the treatment of subependymal giant cell astrocytomas (a brain tumor) and renal AMLs (kidney tumors) in TSC patients. We present monozygotic twin sisters affected with TSC. Since age 4 years, 1 of the sisters has been treated with everolimus; the other sister received no mTOR inhibitor treatment. After 24-month follow-up, everolimus treatment resulted in a significant brain tumor volume decrease in the treated twin. This child presents no facial angiofibroma, and no renal AMLs. The brain tumor in the nontreated sister is stable in size, but in the meantime, she has developed significant facial angiofibroma and renal AMLs. This observation indicates that early mTOR inhibition in TSC patients may prevent the development of TSC lesions and alter the natural history of the disease.

摘要

结节性硬化症复合征(TSC)是一种遗传疾病,其特征是哺乳动物雷帕霉素靶蛋白(mTOR)的激活和良性肿瘤的生长。一些 TSC 病变,如心脏横纹肌瘤和大脑皮质结节,发生在胎儿中,而另一些病变,如肾血管平滑肌脂肪瘤(AML)和皮肤血管纤维瘤,则在数年内发展。最近,mTOR 抑制剂依维莫司被证明对 TSC 患者的室管膜下巨细胞星形细胞瘤(脑肿瘤)和肾 AML(肾肿瘤)的治疗有效。我们报告了一对患有 TSC 的同卵双胞胎姐妹。自 4 岁起,其中一位姐妹接受依维莫司治疗;另一位姐妹未接受 mTOR 抑制剂治疗。经过 24 个月的随访,依维莫司治疗导致治疗组双胞胎的脑肿瘤体积显著减小。该患儿无面部血管纤维瘤,也无肾 AML。未治疗组姐妹的脑肿瘤大小保持稳定,但同时,她出现了明显的面部血管纤维瘤和肾 AML。这一观察表明,早期对 TSC 患者进行 mTOR 抑制可能预防 TSC 病变的发生,并改变疾病的自然病程。

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