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中国儿童科恩氏综合征队列的临床和分子分析。

Clinical and molecular analysis in a cohort of Chinese children with Cornelia de Lange syndrome.

机构信息

Department of Endocrinology and Metabolism, Shanghai Children's Medical Center, Shanghai Jiaotong University School of Medicine, 1678 Dongfang Road, Shanghai, 200127, China.

Department of Rare Disease Clinic, Shanghai Children's Medical Center, Shanghai Jiaotong University School of Medicine, Shanghai, China.

出版信息

Sci Rep. 2020 Dec 4;10(1):21224. doi: 10.1038/s41598-020-78205-5.

Abstract

Cornelia de Lange Syndrome (CdLS) is a rare genetic disorder, which causes a range of physical, cognitive, and medical challenges. To retrospectively analyze the clinical characteristics and genetic variations of Chinese patients, and to provide experience for further diagnosis and treatment of CdLS in Chinese children, we identified 15 unrelated Chinese children who presented with unusual facial features, short stature, developmental delay, limb abnormalities, and a wide range of health conditions. In this study, targeted-next generation sequencing was used to screen for causal variants and the clinically relevant variants were subsequently verified using Sanger sequencing. DNA sequencing identified 15 genetic variations, including 11 NIPBL gene variants, two SMC1A gene variants, one RAD21 gene variant, and one HDAC8 variant. The phenotype of these patients was summarized and differences between this cohort and another four groups were compared. The clinical manifestations of the patients in this cohort were mostly consistent with other ethnicities, but several clinical features in our cohort had different frequencies compared with other groups. We identified 15 deleterious variants of which 11 were novel. Variants in the NIPBL gene were the most common cause in our cohort. Our study not only expands upon the spectrum of genetic variations in CdLS, but also broadens our understanding of the clinical features of CdLS.

摘要

Cornelia de Lange 综合征(CdLS)是一种罕见的遗传性疾病,可导致一系列身体、认知和医学挑战。为了回顾性分析中国患者的临床特征和遗传变异,并为中国儿童 CdLS 的进一步诊断和治疗提供经验,我们鉴定了 15 名无亲缘关系的中国儿童,他们具有异常的面部特征、身材矮小、发育迟缓、肢体异常和广泛的健康状况。在这项研究中,使用靶向下一代测序来筛选致病变异,随后使用 Sanger 测序验证临床相关变异。DNA 测序鉴定了 15 种遗传变异,包括 11 种 NIPBL 基因突变、2 种 SMC1A 基因突变、1 种 RAD21 基因突变和 1 种 HDAC8 突变。总结了这些患者的表型,并比较了本队列与另外四组之间的差异。本队列患者的临床表现与其他种族大多一致,但与其他组相比,本队列的一些临床特征的发生率有所不同。我们鉴定了 15 种有害变异,其中 11 种是新的。在本队列中,NIPBL 基因突变是最常见的病因。本研究不仅扩展了 CdLS 的遗传变异谱,还加深了我们对 CdLS 临床特征的理解。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a886/7718889/2e79e292df7c/41598_2020_78205_Fig1_HTML.jpg

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