• 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

阿曼的翁韦里希特-伦德伯格进行性肌阵挛癫痫

Unverricht-Lundborg progressive myoclonus epilepsy in Oman.

作者信息

Santoshkumar Balagopal, Turnbull Julie, Minassian Berge A

机构信息

Department of Neurology, Royal Hospital, Muscat, Oman.

出版信息

Pediatr Neurol. 2008 Apr;38(4):252-5. doi: 10.1016/j.pediatrneurol.2007.11.006.

DOI:10.1016/j.pediatrneurol.2007.11.006
PMID:18358403
Abstract

We analyzed the clinical, electrophysiologic, and genetic features of Omani Arab patients suspected of manifesting the Unverricht-Lundborg form of progressive myoclonus epilepsy. Ten patients (five boys, five girls; mean age at onset, 10.2 years) were evaluated. Unverricht-Lundborg disease was confirmed in all by detection of dodecamer repeat expansion mutations in the EPM1 gene. There was no correlation between age at onset or severity of disease with sizes of dodecamer repeats. Myoclonic seizures were the presenting symptom in 70% of patients. Myoclonus was severe in adolescence, but remained stable or improved beyond 5-6 years of disease onset. No significant cognitive decline occurred. Nearly 75% of patients exhibited mild to moderate cerebellar dysfunction, which was nonprogressive after adulthood. Slowing of background activity, generalized spike-wave discharges, and photoparoxysmal responses were evident in all patients' electroencephalograms. Spike-wave discharges and photoparoxysmal responses tended to disappear in adulthood. This cluster of progressive myoclonus epilepsy patients manifested typical Unverricht-Lundborg disease. All cases had mutations in EPM1, the known gene for this disorder, and therefore do not contribute to identifying the gene in a second Unverricht-Lundborg disease locus recently mapped in Arab patients from Israel. Although Unverricht-Lundborg disease is very severe in adolescence, its clinical signs stabilize and improve somewhat in adulthood in this so-called "progressive epilepsy."

摘要

我们分析了疑似表现为翁韦里希特-伦德伯格型进行性肌阵挛癫痫的阿曼阿拉伯患者的临床、电生理和遗传特征。对10名患者(5名男孩,5名女孩;平均发病年龄10.2岁)进行了评估。通过检测EPM1基因中的十二聚体重复扩增突变,在所有患者中确诊为翁韦里希特-伦德伯格病。发病年龄或疾病严重程度与十二聚体重复序列的大小之间无相关性。70%的患者以肌阵挛发作作为首发症状。肌阵挛在青春期严重,但在发病5至6年后保持稳定或有所改善。未出现明显的认知功能下降。近75%的患者表现出轻度至中度的小脑功能障碍,成年后无进展。所有患者的脑电图均显示背景活动减慢、广泛性棘波放电和光阵发性反应。棘波放电和光阵发性反应在成年后往往消失。这组进行性肌阵挛癫痫患者表现出典型的翁韦里希特-伦德伯格病。所有病例均在EPM1(该疾病的已知基因)中存在突变,因此对于在最近从以色列阿拉伯患者中定位的第二个翁韦里希特-伦德伯格病基因座中鉴定基因没有帮助。尽管翁韦里希特-伦德伯格病在青春期非常严重,但其临床症状在成年期会在这种所谓的“进行性癫痫”中有所稳定和改善。

相似文献

1
Unverricht-Lundborg progressive myoclonus epilepsy in Oman.阿曼的翁韦里希特-伦德伯格进行性肌阵挛癫痫
Pediatr Neurol. 2008 Apr;38(4):252-5. doi: 10.1016/j.pediatrneurol.2007.11.006.
2
A new clinical and molecular form of Unverricht-Lundborg disease localized by homozygosity mapping.通过纯合性定位确定的一种新的临床和分子形式的翁韦里希特-伦德伯格病
Brain. 2005 Mar;128(Pt 3):652-8. doi: 10.1093/brain/awh377. Epub 2005 Jan 5.
3
Clinical picture of EPM1-Unverricht-Lundborg disease.EPM1型——翁韦里希特-伦德伯格病的临床表现。
Epilepsia. 2008 Apr;49(4):549-56. doi: 10.1111/j.1528-1167.2008.01546.x. Epub 2008 Mar 5.
4
Cystatin B: mutation detection, alternative splicing and expression in progressive myclonus epilepsy of Unverricht-Lundborg type (EPM1) patients.胱抑素B:翁韦里希特-伦德伯格型进行性肌阵挛癫痫(EPM1)患者中的突变检测、可变剪接及表达
Eur J Hum Genet. 2007 Feb;15(2):185-93. doi: 10.1038/sj.ejhg.5201723. Epub 2006 Sep 27.
5
Severer phenotype in Unverricht-Lundborg disease (EPM1) patients compound heterozygous for the dodecamer repeat expansion and the c.202C>T mutation in the CSTB gene.Unverricht-Lundborg 病(EPM1)患者复合杂合 CSTB 基因的十二聚体重复扩展和 c.202C>T 突变表现出更严重的表型。
Neurodegener Dis. 2011;8(6):515-22. doi: 10.1159/000323470. Epub 2011 Jul 15.
6
Molecular background of EPM1-Unverricht-Lundborg disease.EPM1型翁韦里希特-伦德伯格病的分子背景
Epilepsia. 2008 Apr;49(4):557-63. doi: 10.1111/j.1528-1167.2007.01422.x. Epub 2007 Nov 19.
7
[From gene to disease; progressive myoclonus epilepsy of Unverricht-Lundborg and mutations in the cystatin B gene].[从基因到疾病;翁韦里希特-伦德伯格进行性肌阵挛性癫痫与胱抑素B基因突变]
Ned Tijdschr Geneeskd. 2002 May 4;146(18):846-8.
8
DNA deamination enables direct PCR amplification of the cystatin B (CSTB) gene-associated dodecamer repeat expansion in myoclonus epilepsy type Unverricht-Lundborg.DNA脱氨基作用可实现对昂韦里希特-伦德伯格型肌阵挛性癫痫中胱抑素B(CSTB)基因相关的十二聚体重复序列扩增进行直接PCR扩增。
Hum Mutat. 2003 Nov;22(5):404-8. doi: 10.1002/humu.10276.
9
Electroclinical presentation and genotype-phenotype relationships in patients with Unverricht-Lundborg disease carrying compound heterozygous CSTB point and indel mutations.伴有复合杂合 CSTB 点突变和缺失突变的 Unverricht-Lundborg 病患者的电临床表型和基因型-表型关系。
Epilepsia. 2012 Dec;53(12):2120-7. doi: 10.1111/j.1528-1167.2012.03718.x.
10
Unverricht-Lundborg disease in a five-generation Arab family: instability of dodecamer repeats.一个五代阿拉伯家族中的翁韦里希特-伦德伯格病:十二聚体重复序列的不稳定性
Neurology. 2001 Sep 25;57(6):1050-4. doi: 10.1212/wnl.57.6.1050.

引用本文的文献

1
Autistic features in Unverricht-Lundborg disease.翁韦里希特-伦德伯格病中的自闭症特征。
Epilepsy Behav Rep. 2019 Jun 6;12:100323. doi: 10.1016/j.ebr.2019.100323. eCollection 2019.
2
Progressive volume loss and white matter degeneration in cstb-deficient mice: a diffusion tensor and longitudinal volumetry MRI study.Cstb基因缺陷小鼠的进行性体积损失和白质变性:一项扩散张量和纵向容积MRI研究。
PLoS One. 2014 Mar 6;9(6):e90709. doi: 10.1371/journal.pone.0090709. eCollection 2014.
3
3D texture analysis reveals imperceptible MRI textural alterations in the thalamus and putamen in progressive myoclonic epilepsy type 1, EPM1.
3D 纹理分析显示进行性肌阵挛性癫痫 1 型(EPM1)患者丘脑和壳核的 MRI 纹理改变难以察觉。
PLoS One. 2013 Jul 29;8(7):e69905. doi: 10.1371/journal.pone.0069905. Print 2013.
4
Sensorimotor, visual, and auditory cortical atrophy in Unverricht-Lundborg disease mapped with cortical thickness analysis.皮质厚度分析显示,Unverricht-Lundborg 病患者存在感觉运动、视觉和听觉皮质萎缩。
AJNR Am J Neuroradiol. 2012 May;33(5):878-83. doi: 10.3174/ajnr.A2882. Epub 2012 Jan 19.