• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

印度雷特综合征患者的遗传不稳定性与疾病进展。

Genetic Instability and Disease Progression of Indian Rett Syndrome Patients.

机构信息

Centre for Neuroscience, Department of Biotechnology, Karpagam Academy of Higher Education (Deemed to be University), Coimbatore, Tamil Nadu, 641021, India.

Department of Human Genetics and Molecular Biology, Bharathiar University, Coimbatore, Tamil Nadu, 641 046, India.

出版信息

Mol Neurobiol. 2024 Jul;61(7):4868-4878. doi: 10.1007/s12035-023-03882-y. Epub 2023 Dec 26.

DOI:10.1007/s12035-023-03882-y
PMID:38147229
Abstract

Rett syndrome (RTT) is the rare neurodevelopmental disorder caused by mutations in methyl CpG binding protein 2 (MECP2) gene with a prevalence of 1:10,000 worldwide. The hallmark clinical features of RTT are developmental delay, microcephaly, repetitive behaviours, gait abnormalities, respiratory abnormalities and seizures. Still, the understanding on the diagnosis of RTT among clinicians are less. The aim of our work was to study various clinical manifestations and a spectrum of MECP2 genetic heterogeneity in RTT patients from South Indian population. We screened 208 autistic patients and diagnosed 20 RTT patients, who were further divided into classical RTT (group I; N = 11) and variant RTT (group II; N = 9). The clinical severity of RTT was measured using RSSS, RSBQ, SSI, SSS and RTT gross motor scale. The biochemical analysis showed that thyroid-stimulating hormone (TSH), plasma dopamine and cholesterol levels were higher in group I when compared to group II, whereas the level of blood pressure, calcium, ferritin and high-density lipoprotein levels were significantly decreased in both RTT groups, when compared to the control group. The genetic mutational spectrum of MECP2 mutations were found in 12/20 of RTT patients, which revealed the occurrence of 60% pathogenic mutation and 20% unknown mutation and it was correlated with the clinical finding of respiratory dysfunction, scoliosis and sleeping problems. The significant results of this study provided clinical and genetic aspects of RTT diagnosis and proposed the clinicians to screen abnormal cholesterol, calcium and TSH levels tailed with MECP2 gene mutations for early prognosis of disease severity.

摘要

雷特综合征(RTT)是一种罕见的神经发育障碍,由甲基 CpG 结合蛋白 2(MECP2)基因突变引起,全球患病率为 1:10000。RTT 的标志性临床特征是发育迟缓、小头畸形、重复行为、步态异常、呼吸异常和癫痫发作。然而,临床医生对 RTT 的诊断理解仍较少。我们的工作旨在研究来自印度南部人群的 RTT 患者的各种临床表现和 MECP2 基因异质性谱。我们筛选了 208 名自闭症患者,诊断出 20 名 RTT 患者,其中进一步分为经典 RTT(I 组;N = 11)和变异 RTT(II 组;N = 9)。使用 RSSS、RSBQ、SSI、SSS 和 RTT 粗大运动量表测量 RTT 的临床严重程度。生化分析显示,与 II 组相比,I 组的促甲状腺激素(TSH)、血浆多巴胺和胆固醇水平较高,而两组 RTT 的血压、钙、铁蛋白和高密度脂蛋白水平均显著降低,与对照组相比。在 20 名 RTT 患者中发现了 MECP2 突变的遗传突变谱,其中 60%的致病性突变和 20%的未知突变与呼吸功能障碍、脊柱侧凸和睡眠问题的临床发现相关。这项研究的重要结果提供了 RTT 诊断的临床和遗传方面,并提出临床医生筛选异常胆固醇、钙和 TSH 水平以及 MECP2 基因突变,以早期预测疾病严重程度。

相似文献

1
Genetic Instability and Disease Progression of Indian Rett Syndrome Patients.印度雷特综合征患者的遗传不稳定性与疾病进展。
Mol Neurobiol. 2024 Jul;61(7):4868-4878. doi: 10.1007/s12035-023-03882-y. Epub 2023 Dec 26.
2
Social impairments in Rett syndrome: characteristics and relationship with clinical severity.雷特综合征的社交障碍:特征及与临床严重程度的关系。
J Intellect Disabil Res. 2012 Mar;56(3):233-47. doi: 10.1111/j.1365-2788.2011.01404.x. Epub 2011 Mar 8.
3
Mutational analysis of methyl-CpG binding protein 2 (MECP2) gene in Indian cases of Rett syndrome.对印度雷特综合征病例中甲基化CpG 结合蛋白 2 (MECP2) 基因的突变分析。
J Clin Lab Anal. 2013 Mar;27(2):137-42. doi: 10.1002/jcla.21574. Epub 2013 Feb 11.
4
Methyl-CpG-binding protein 2 (MECP2) mutation type is associated with disease severity in Rett syndrome.甲基化CpG 结合蛋白 2(MECP2)突变类型与雷特综合征的疾病严重程度相关。
J Med Genet. 2014 Mar;51(3):152-8. doi: 10.1136/jmedgenet-2013-102113. Epub 2014 Jan 7.
5
MECP2 mutations and clinical correlations in Greek children with Rett syndrome and associated neurodevelopmental disorders.希腊患有雷特综合征及相关神经发育障碍儿童的MECP2突变与临床相关性
Brain Dev. 2012 Jun;34(6):487-95. doi: 10.1016/j.braindev.2011.09.002. Epub 2011 Oct 6.
6
MECP2 mutations in Czech patients with Rett syndrome and Rett-like phenotypes: novel mutations, genotype-phenotype correlations and validation of high-resolution melting analysis for mutation scanning.捷克雷特综合征及类雷特综合征表型患者的MECP2突变:新突变、基因型-表型相关性及用于突变扫描的高分辨率熔解分析的验证
J Hum Genet. 2016 Jul;61(7):617-25. doi: 10.1038/jhg.2016.19. Epub 2016 Mar 17.
7
Genotype-phenotype relationship among Egyptian children with Rett syndrome.埃及雷特综合征患儿的基因型与表型关系
J Egypt Public Health Assoc. 2015 Sep;90(3):133-7. doi: 10.1097/01.EPX.0000469901.73624.7a.
8
[Genetic features and mechanism of Rett syndrome in Chinese population].[中国人群中雷特综合征的遗传特征及机制]
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2014 Feb;31(1):1-5. doi: 10.3760/cma.j.issn.1003-9406.2014.01.001.
9
Methyl-CpG-binding protein 2 (MECP2) mutation type is associated with bone disease severity in Rett syndrome.甲基化CpG 结合蛋白 2(MECP2)突变类型与 Rett 综合征骨病严重程度相关。
BMC Med Genet. 2020 Jan 31;21(1):21. doi: 10.1186/s12881-020-0960-2.
10
Surface- and voxel-based brain morphologic study in Rett and Rett-like syndrome with MECP2 mutation.基于表面和体素的雷特综合征及类雷特综合征伴MECP2突变的脑形态学研究。
Int J Dev Neurosci. 2019 Apr;73:83-88. doi: 10.1016/j.ijdevneu.2019.01.005. Epub 2019 Jan 25.

本文引用的文献

1
Highlighting novel genes associated with the classical Rett syndrome patient from India.突出显示与来自印度的典型雷特综合征患者相关的新基因。
Genes Dis. 2022 Jan 6;9(6):1394-1396. doi: 10.1016/j.gendis.2021.12.002. eCollection 2022 Nov.
2
Role of DNA Methyl-CpG-Binding Protein MeCP2 in Rett Syndrome Pathobiology and Mechanism of Disease.DNA 甲基-CpG 结合蛋白 MeCP2 在 Rett 综合征发病机制中的作用。
Biomolecules. 2021 Jan 8;11(1):75. doi: 10.3390/biom11010075.
3
Drug Studies on Rett Syndrome: From Bench to Bedside.雷特综合征的药物研究:从基础到临床。
J Autism Dev Disord. 2020 Aug;50(8):2740-2764. doi: 10.1007/s10803-020-04381-y.
4
Autism in India: a case-control study to understand the association between socio-economic and environmental risk factors.印度自闭症病例对照研究:了解社会经济和环境风险因素之间的关联。
Acta Neurol Belg. 2019 Sep;119(3):393-401. doi: 10.1007/s13760-018-01057-4. Epub 2018 Dec 15.
5
Monogenic diseases in India.印度的单基因疾病。
Mutat Res Rev Mutat Res. 2018 Apr-Jun;776:23-31. doi: 10.1016/j.mrrev.2018.03.003. Epub 2018 Mar 17.
6
Epigenetic Etiology of Intellectual Disability.智力残疾的表观遗传学病因
J Neurosci. 2017 Nov 8;37(45):10773-10782. doi: 10.1523/JNEUROSCI.1840-17.2017.
7
A review of Rett syndrome (RTT) with induced pluripotent stem cells.诱导多能干细胞对雷特综合征(RTT)的综述。
Stem Cell Investig. 2016 Sep 28;3:52. doi: 10.21037/sci.2016.09.05. eCollection 2016.
8
Blood oxidative stress and metallothionein expression in Rett syndrome: Probing for markers.
World J Biol Psychiatry. 2016 Apr;17(3):198-209. doi: 10.3109/15622975.2015.1077990. Epub 2015 Oct 15.
9
Thyroid function in Rett syndrome.雷特综合征中的甲状腺功能。
Horm Res Paediatr. 2015;83(2):118-25. doi: 10.1159/000370066. Epub 2015 Jan 21.
10
Rett syndrome and MeCP2.雷特综合征与 MeCP2。
Neuromolecular Med. 2014 Jun;16(2):231-64. doi: 10.1007/s12017-014-8295-9. Epub 2014 Mar 11.