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早发性发育性和癫痫性脑病的表型和基因型谱 - 来自罗马尼亚队列的数据。

Phenotypic and Genotypic Spectrum of Early-Onset Developmental and Epileptic Encephalopathies-Data from a Romanian Cohort.

机构信息

Regional Centre of Medical Genetics Dolj, Emergency County Hospital Craiova, 200642 Craiova, Romania.

Laboratory of Human Genomics, University of Medicine and Pharmacy of Craiova, 200638 Craiova, Romania.

出版信息

Genes (Basel). 2022 Jul 15;13(7):1253. doi: 10.3390/genes13071253.

Abstract

Early-onset developmental epileptic encephalopathy (DEE) refers to an age-specific, diverse group of epilepsy syndromes with electroclinical anomalies that are associated with severe cognitive, behavioral, and developmental impairments. Genetic DEEs have heterogeneous etiologies. This study includes 36 Romanian patients referred to the Regional Centre for Medical Genetics Dolj for genetic testing between 2017 and 2020. The patients had been admitted to and clinically evaluated at Doctor Victor Gomoiu Children’s Hospital and Prof. Dr. Alexandru Obregia Psychiatry Hospital in Bucharest. Panel testing was performed using the Illumina® TruSight™ One “clinical exome” (4811 genes), and the analysis focused on the known genes reported in DEEs and clinical concordance. The overall diagnostic rate was 25% (9/36 cases). Seven cases were diagnosed with Dravet syndrome (likely pathogenic/pathogenic variants in SCN1A) and two with Genetic Epilepsy with Febrile Seizures Plus (SCN1B). For the diagnosed patients, seizure onset was <1 year, and the seizure type was generalized tonic-clonic. Four additional plausible variants of unknown significance in SCN2A, SCN9A, and SLC2A1 correlated with the reported phenotype. Overall, we are reporting seven novel variants. Comprehensive clinical phenotyping is crucial for variant interpretation. Genetic assessment of patients with severe early-onset DEE can be a powerful diagnostic tool for clinicians, with implications for the management and counseling of the patients and their families.

摘要

早发性发育性癫痫性脑病 (DEE) 是指一种特定于年龄的、具有电临床异常的癫痫综合征的多样化群体,与严重的认知、行为和发育障碍有关。遗传性 DEE 的病因具有异质性。本研究纳入了 36 名罗马尼亚患者,他们于 2017 年至 2020 年期间在多尔日地区医学遗传学中心接受基因检测。这些患者曾在布加勒斯特的 Victor Gomoiu 儿童医院和 Alexandru Obregia 精神病医院接受过住院治疗和临床评估。采用 Illumina® TruSight™ One“临床外显子组”(4811 个基因)进行面板检测,分析重点为 DEE 和临床一致性相关的已知基因。总体诊断率为 25%(36 例中的 9 例)。7 例诊断为 Dravet 综合征(SCN1A 中可能致病/致病性变异),2 例诊断为伴热性惊厥附加症的遗传性癫痫(SCN1B)。对于已确诊的患者,癫痫发作起始于<1 岁,发作类型为全面强直阵挛性发作。SCN2A、SCN9A 和 SLC2A1 中另外 4 种可能具有未知意义的变异与报告的表型相关。总体而言,我们报告了 7 种新的变异。全面的临床表型分析对于变异解释至关重要。对严重早发性 DEE 患者进行基因评估可以为临床医生提供一种强大的诊断工具,对患者及其家属的管理和咨询具有重要意义。

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本文引用的文献

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Dravet syndrome.德拉韦综合征。
Curr Opin Neurol. 2021 Apr 1;34(2):213-218. doi: 10.1097/WCO.0000000000000902.
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Genetic Testing in Epilepsy.癫痫的基因检测
Semin Neurol. 2020 Dec;40(6):730-738. doi: 10.1055/s-0040-1719070. Epub 2020 Nov 11.

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