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中国人群中伴有 NSD1 突变的索托斯综合征:两种新突变的鉴定及文献综述

Sotos Syndrome With NSD1 Mutations in a Chinese Cohort: Identification of Two Novel Mutations and Literature Review.

作者信息

Zhao Jianhui, Li Luzhuang, Sun Dianrong, Zhang Leihong, Liu Lin, Hou Mei

机构信息

Department of Neurology & Rehabilitation, Qingdao Women and Children's Hospital, Qingdao University, Qingdao, Shandong, People's Republic of China.

出版信息

Int J Dev Neurosci. 2025 Aug;85(5):e70032. doi: 10.1002/jdn.70032.

DOI:10.1002/jdn.70032
PMID:40693312
Abstract

Sotos syndrome is an autosomal dominant disorder resulting from pathogenic variants of the NSD1 gene. In this study, we present five Chinese paediatric cases, including two previously unreported NSD1 variants: a nonsense mutation (c.1486A > T p. Lys496*) and a missense mutation (c.6086C > T) (p. Thr2029Ile) respectively. Additionally, we analyzed the genotypic and phenotypic spectrum of 23 Chinese children with molecularly confirmed Sotos syndrome. Patients exhibited characteristic craniofacial features and significant overgrowth. All patients showed DD/ID and five patients (21.7%) showed symptoms of ASD. Febrile seizures occurred in six patients (26.1%). Abnormalities on cranial imaging were generally nonspecific. Other clinical features were also shown, such as atrial septal defect (5 cases), patent ductus arteriosus (3 cases), scoliosis (2 cases) and neonatal hypoglycemia (2 cases). These findings underscore the phenotypic variability of Sotos syndrome and highlight the necessity for long-term multidisciplinary follow-up to delineate its evolving natural history and optimize clinical management.

摘要

索托斯综合征是一种由NSD1基因的致病变异引起的常染色体显性疾病。在本研究中,我们报告了5例中国儿科病例,包括2个此前未报道的NSD1变异:分别为一个无义突变(c.1486A>T,p.Lys496*)和一个错义突变(c.6086C>T)(p.Thr2029Ile)。此外,我们分析了23例经分子学确诊的中国索托斯综合征患儿的基因型和表型谱。患者表现出典型的颅面特征和显著的过度生长。所有患者均有发育迟缓/智力障碍,5例患者(21.7%)表现出自闭症谱系障碍症状。6例患者(26.1%)发生热性惊厥。头颅影像学异常通常不具有特异性。还表现出其他临床特征,如房间隔缺损(5例)、动脉导管未闭(3例)、脊柱侧弯(2例)和新生儿低血糖(2例)。这些发现强调了索托斯综合征的表型变异性,并突出了长期多学科随访以描绘其不断演变的自然病史和优化临床管理的必要性。

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本文引用的文献

1
Identification of Novel NSD1 variations in four Pediatric cases with sotos Syndrome.在四个 Sotos 综合征儿科病例中鉴定出新型 NSD1 变异。
BMC Med Genomics. 2024 Apr 29;17(1):116. doi: 10.1186/s12920-024-01889-5.
2
The first pineoblastoma case report of a patient with Sotos syndrome harboring NSD1 germline mutation.首例 Sotos 综合征伴 NSD1 种系突变的松果体母细胞瘤病例报告。
BMC Pediatr. 2024 Mar 8;24(1):166. doi: 10.1186/s12887-024-04636-y.
3
Sotos syndrome treated with traditional Chinese medicine and rehabilitation: Case report.
采用中医药结合康复治疗 Sotos 综合征 1 例报告。
Medicine (Baltimore). 2023 Dec 1;102(48):e36169. doi: 10.1097/MD.0000000000036169.
4
A novel nonsense variant in NSD1 gene in a female child with Sotos syndrome: A case report and literature review.一个患有 Sotos 综合征的女性患儿 NSD1 基因中一个新的无义变异:病例报告及文献复习。
Brain Behav. 2023 Dec;13(12):e3290. doi: 10.1002/brb3.3290. Epub 2023 Oct 31.
5
Case Report of Neonatal Sotos Syndrome with a New Missense Mutation in the Gene and Literature Analysis in the Chinese Han Population.新生儿 Sotos 综合征病例报告,该基因中存在新的错义突变,并对中国汉族人群进行文献分析。
Medicina (Kaunas). 2022 Jul 21;58(7):968. doi: 10.3390/medicina58070968.
6
RNA Analysis and Clinical Characterization of a Novel Splice Variant in the Gene Causing Familial Sotos Syndrome.导致家族性索托斯综合征的基因中一种新型剪接变体的RNA分析及临床特征
Front Pediatr. 2022 Feb 2;10:827802. doi: 10.3389/fped.2022.827802. eCollection 2022.
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[Genetic analysis of a child with Sotos syndrome].
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2020 Feb 10;37(2):127-130. doi: 10.3760/cma.j.issn.1003-9406.2020.02.006.
8
DNA methylation analysis of multiple imprinted DMRs in Sotos syndrome reveals IGF2-DMR0 as a DNA methylation-dependent, P0 promoter-specific enhancer.多重印记 DMRs 的 DNA 甲基化分析揭示 IGF2-DMR0 是一个 DNA 甲基化依赖的、P0 启动子特异性增强子。
FASEB J. 2020 Jan;34(1):960-973. doi: 10.1096/fj.201901757R. Epub 2019 Nov 28.
9
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Clin Cancer Res. 2017 Jun 15;23(12):e83-e90. doi: 10.1158/1078-0432.CCR-17-0631.
10
The Role of Nuclear Receptor-Binding SET Domain Family Histone Lysine Methyltransferases in Cancer.核受体结合SET结构域家族组蛋白赖氨酸甲基转移酶在癌症中的作用
Cold Spring Harb Perspect Med. 2017 Jun 1;7(6):a026708. doi: 10.1101/cshperspect.a026708.