结节性硬化症的遗传病因、诊断和治疗。

IF 7.7 2区 生物学 Q1 GENETICS & HEREDITY Annual review of genomics and human genetics Pub Date : 2019-08-31 Epub Date: 2019-04-24 DOI:10.1146/annurev-genom-083118-015354
Catherine L Salussolia, Katarzyna Klonowska, David J Kwiatkowski, Mustafa Sahin
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引用次数: 86

摘要

结节性硬化症(TSC)是一种常染色体显性疾病,由于TSC1或TSC2失活变异,导致雷帕霉素(mTOR)通路的机制靶点过度激活,影响多器官系统。失调的mTOR信号导致细胞生长和增殖增加。临床上,TSC患者表现出很大的表型变异性,但疾病的神经和神经精神表现具有最高的发病率和死亡率。tsc相关性癫痫几乎发生在所有患者中,并且由于多种抗癫痫药物难以治疗,通常难以治疗。mTOR抑制剂的出现为治疗tsc相关癫痫和该疾病的其他神经发育表现提供了巨大的希望;然而,治疗干预的最佳时机尚不完全清楚。
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Genetic Etiologies, Diagnosis, and Treatment of Tuberous Sclerosis Complex.

Tuberous sclerosis complex (TSC) is an autosomal dominant disorder that affects multiple organ systems due to an inactivating variant in either TSC1 or TSC2, resulting in the hyperactivation of the mechanistic target of rapamycin (mTOR) pathway. Dysregulated mTOR signaling results in increased cell growth and proliferation. Clinically, TSC patients exhibit great phenotypic variability, but the neurologic and neuropsychiatric manifestations of the disease have the greatest morbidity and mortality. TSC-associated epilepsy occurs in nearly all patients and is often difficult to treat because it is refractory to multiple antiseizure medications. The advent of mTOR inhibitors offers great promise in the treatment of TSC-associated epilepsy and other neurodevelopmental manifestations of the disease; however, the optimal timing of therapeutic intervention is not yet fully understood.

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来源期刊
CiteScore
14.90
自引率
1.10%
发文量
29
期刊介绍: Since its inception in 2000, the Annual Review of Genomics and Human Genetics has been dedicated to showcasing significant developments in genomics as they pertain to human genetics and the human genome. The journal emphasizes genomic technology, genome structure and function, genetic modification, human variation and population genetics, human evolution, and various aspects of human genetic diseases, including individualized medicine.
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