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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.

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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