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扩展与ARID2相关的智力残疾的临床和遗传谱。7例患者的病例系列。

Extending the clinical and genetic spectrum of ARID2 related intellectual disability. A case series of 7 patients.

作者信息

Gazdagh Gabriella, Blyth Moira, Scurr Ingrid, Turnpenny Peter D, Mehta Sarju G, Armstrong Ruth, McEntagart Meriel, Newbury-Ecob Ruth, Tobias Edward S, Joss Shelagh

机构信息

West of Scotland Regional Genetics Service, Laboratory Medicine Building, Queen Elizabeth University Hospital, Glasgow, United Kingdom.

Yorkshire Regional Genetics Service, Department of Clinical Genetics, Chapel Allerton Hospital, Leeds, United Kingdom.

出版信息

Eur J Med Genet. 2019 Jan;62(1):27-34. doi: 10.1016/j.ejmg.2018.04.014. Epub 2018 Apr 23.

DOI:10.1016/j.ejmg.2018.04.014
PMID:29698805
Abstract

In the last 3 years de novo sequence variants in the ARID2 (AT-rich interaction domain 2) gene, a subunit of the SWI/SNF complex, have been linked to intellectual disabilities in 3 case reports including one which describes frameshift mutations in ARID2 in 2 patients with features resembling Coffin-Siris syndrome. Coffin-Siris syndrome (CSS) is a rare congenital syndrome characterized by intellectual deficit, coarse facial features and hypoplastic or absent fifth fingernails and/or toenails among other features. Mutations in a number of different genes encoding SWI/SNF chromatin remodelling complex proteins have been described but the underlying molecular cause remains unknown in approximately 40% of patients with CSS. Here we describe 7 unrelated individuals, 2 with deletions of the ARID2 region and 5 with de novo truncating mutations in the ARID2 gene. Similarities to CSS are evident. Although hypertrichosis and hypoplasia of the fifth finger nail and distal phalanx do not appear to be common in these patients, toenail hypoplasia and the presence of Wormian bones might support the involvement of ARID2.

摘要

在过去3年中,SWI/SNF复合物的一个亚基——ARID2(富含AT交互结构域2)基因中的新生序列变异,在3例病例报告中与智力残疾相关,其中1例描述了2例具有类似科芬-西里斯综合征特征患者的ARID2移码突变。科芬-西里斯综合征(CSS)是一种罕见的先天性综合征,其特征包括智力缺陷、面部特征粗糙、第五指(趾)甲发育不全或缺失等。已经描述了许多编码SWI/SNF染色质重塑复合蛋白的不同基因中的突变,但在大约40%的CSS患者中,潜在的分子病因仍然未知。在此,我们描述了7名无亲缘关系的个体,其中2名ARID2区域存在缺失,5名ARID2基因发生新生截断突变。与CSS的相似性很明显。尽管多毛症以及第五指(趾)甲和远端指骨发育不全在这些患者中似乎并不常见,但趾甲发育不全和缝间骨的存在可能支持ARID2与之相关。

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Frameshift Variant in in a Chilean Individual with Coffin-Siris Syndrome Phenotype.一名具有科芬-西里斯综合征表型的智利个体中的移码变异
J Pediatr Genet. 2021 Dec 24;13(2):149-153. doi: 10.1055/s-0041-1740531. eCollection 2024 Jun.
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Front Pediatr. 2022 Jun 23;10:911954. doi: 10.3389/fped.2022.911954. eCollection 2022.
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