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结节性硬化症的终身管理的护理意义。

Nursing implications for the lifelong management of tuberous sclerosis complex.

机构信息

Tuberous Sclerosis Clinic, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.

出版信息

J Neurosci Nurs. 2013 Aug;45(4):226-42. doi: 10.1097/JNN.0b013e3182986146.

Abstract

Tuberous sclerosis complex (TSC) is a genetic disorder that can affect multiple organ systems, including the brain, heart, skin, kidney, and lung, by formation of benign hamartomas. It can be associated with autism, epilepsy, and other neurocognitive and behavioral disabilities. The incidence of TSC is approximately 1 in 6,000 live births, but it may be underdiagnosed. Mutations to either the TSC1 (coding for hamartin) or TSC2 (coding for tuberin) genes are present in 85% of patients with TSC. The TSC1/TSC2 protein dimer complex is a crucial inhibitory element in the mammalian target of rapamycin (mTOR) complex 1 pathway that regulates cell growth and proliferation. The manifestations of TSC usually require management over the entire life of the patient. Until recently, there were few options, other than surgical removal, for treating the symptoms of TSC related to growth of hamartomas. Increased understanding of the genetic cause of the disease and the underlying dysregulation of the mTOR pathway has led to clinical trials of mTOR inhibitors including sirolimus and everolimus. This article will review the various manifestations of TSC and describe treatment strategies, recommendations for surveillance, and use of mTOR inhibitors in their management.

摘要

结节性硬化症复合征(TSC)是一种遗传性疾病,可通过良性错构瘤的形成影响多个器官系统,包括大脑、心脏、皮肤、肾脏和肺。它可伴有自闭症、癫痫和其他神经认知及行为障碍。TSC 的发病率约为每 6000 例活产儿中有 1 例,但可能诊断不足。85%的 TSC 患者存在 TSC1(编码错构素)或 TSC2(编码结节素)基因突变。TSC1/TSC2 蛋白二聚体复合物是哺乳动物雷帕霉素靶蛋白(mTOR)复合物 1 通路中一个重要的抑制性元件,调节细胞生长和增殖。TSC 的表现通常需要在患者的整个生命周期内进行管理。直到最近,除了手术切除外,针对 TSC 相关错构瘤生长的症状,治疗方法选择非常有限。对疾病遗传原因和 mTOR 通路失调的深入了解,导致了雷帕霉素及其衍生物依维莫司等 mTOR 抑制剂的临床试验。本文将综述 TSC 的各种表现,并描述治疗策略、监测建议以及在其治疗中的 mTOR 抑制剂的应用。

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