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技术进步在雷特综合征谱系障碍的基因诊断中的应用。

Technological Improvements in the Genetic Diagnosis of Rett Syndrome Spectrum Disorders.

机构信息

Fundació per la Recerca Sant Joan de Déu, Santa Rosa 39-57, 08950 Esplugues de Llobregat, Spain.

Institut de Recerca Sant Joan de Déu, Santa Rosa 39-57, 08950 Esplugues de Llobregat, Spain.

出版信息

Int J Mol Sci. 2021 Sep 26;22(19):10375. doi: 10.3390/ijms221910375.

Abstract

Rett syndrome (RTT) is a severe neurodevelopmental disorder that constitutes the second most common cause of intellectual disability in females worldwide. In the past few years, the advancements in genetic diagnosis brought by next generation sequencing (NGS), have made it possible to identify more than 90 causative genes for RTT and significantly overlapping phenotypes (RTT spectrum disorders). Therefore, the clinical entity known as RTT is evolving towards a spectrum of overlapping phenotypes with great genetic heterogeneity. Hence, simultaneous multiple gene testing and thorough phenotypic characterization are mandatory to achieve a fast and accurate genetic diagnosis. In this review, we revise the evolution of the diagnostic process of RTT spectrum disorders in the past decades, and we discuss the effectiveness of state-of-the-art genetic testing options, such as clinical exome sequencing and whole exome sequencing. Moreover, we introduce recent technological advancements that will very soon contribute to the increase in diagnostic yield in patients with RTT spectrum disorders. Techniques such as whole genome sequencing, integration of data from several "omics", and mosaicism assessment will provide the tools for the detection and interpretation of genomic variants that will not only increase the diagnostic yield but also widen knowledge about the pathophysiology of these disorders.

摘要

雷特综合征(RTT)是一种严重的神经发育障碍,是全球女性智力残疾的第二大常见原因。在过去的几年中,下一代测序(NGS)带来的遗传诊断进步使得鉴定 RTT 及其显著重叠表型(RTT 谱障碍)的 90 多个致病基因成为可能。因此,被称为 RTT 的临床实体正在朝着具有高度遗传异质性的重叠表型演变。因此,同时进行多个基因检测和全面的表型特征描述对于实现快速、准确的遗传诊断是强制性的。在这篇综述中,我们回顾了过去几十年 RTT 谱障碍诊断过程的演变,并讨论了最先进的遗传检测选择的有效性,如临床外显子组测序和全外显子组测序。此外,我们还介绍了最近的技术进步,这些进步将很快有助于提高 RTT 谱障碍患者的诊断率。全基因组测序、来自多个“组学”的数据整合以及镶嵌性评估等技术将为检测和解释基因组变异提供工具,这不仅将提高诊断率,还将拓宽对这些疾病病理生理学的认识。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/911c/8508637/3d4957574514/ijms-22-10375-g001.jpg

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