脆性X染色体综合征:经验教训和新的治疗途径即将出现。

IF 11.2 1区 医学 Q1 PHARMACOLOGY & PHARMACY
Randi J Hagerman, Paul J Hagerman
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引用次数: 4

摘要

脆性X染色体综合征(FXS)是最常见的遗传性智力残疾,也是自闭症谱系障碍的主要单基因形式,包括认知、行为和身体的临床表现。FXS是由FMR1基因中的非编码CGG重复序列(>200重复)大量扩增引起的,此时该基因通常处于沉默状态。FMR1蛋白(FMRP)对突触的发育和维持至关重要,其缺失会导致神经发育障碍。目前还没有一种治疗方法可以直接逆转FMRP的丧失;然而,针对FXS中失调通路的潜在治疗方法越来越多,包括解决mGluR5通路活性增强和GABA通路缺陷的治疗方法。根据迄今为止的靶向治疗研究,在不久的将来,FXS的一种或多种有效治疗方法的前景良好。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Fragile X Syndrome: Lessons Learned and What New Treatment Avenues Are on the Horizon.

Fragile X syndrome (FXS) is the most common form of inherited intellectual disability and the leading single-gene form of autism spectrum disorder, encompassing cognitive, behavioral, and physical forms of clinical involvement. FXS is caused by large expansions of a noncoding CGG repeat (>200 repeats) in the FMR1 gene, at which point the gene is generally silenced. Absence of FMR1 protein (FMRP), important for synaptic development and maintenance, gives rise to the neurodevelopmental disorder. There is, at present, no therapeutic approach that directly reverses the loss of FMRP; however, there is an increasing number of potential treatments that target the pathways dysregulated in FXS, including those that address the enhanced activity of the mGluR5 pathway and deficits in GABA pathways. Based on studies of targeted therapeutics to date, the prospects are good for one or more effective therapies for FXS in the near future.

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来源期刊
CiteScore
27.80
自引率
0.00%
发文量
53
期刊介绍: Since 1961, the Annual Review of Pharmacology and Toxicology has been a comprehensive resource covering significant developments in pharmacology and toxicology. The journal encompasses various aspects, including receptors, transporters, enzymes, chemical agents, drug development science, and systems like the immune, nervous, gastrointestinal, cardiovascular, endocrine, and pulmonary systems. Special topics are also featured in this annual review.
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