• 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

儿童消失性白质病:一种不寻常的关联、一种新突变及文献综述

Vanishing White Matter Disease in Children: An Unusual Association, a Novel Mutation, and a Literature Review.

作者信息

Alsahlawi Zahra, Isa Hasan M, Alresias Sulaiman, Hasan Sayed Mohamed, Malalla Husain A, Ebrahim Ayman K, Ali Khadija

机构信息

Department of Pediatrics, Arabian Gulf University, Manama, BHR.

Department of Pediatrics, Salmaniya Medical Complex, Manama, BHR.

出版信息

Cureus. 2024 Nov 14;16(11):e73667. doi: 10.7759/cureus.73667. eCollection 2024 Nov.

DOI:10.7759/cureus.73667
PMID:39677130
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11645671/
Abstract

Vanishing white matter (VWM) disease is an autosomal recessive disorder caused by mutations in the gene EIF2B encoding the subunits 1-5 of eukaryotic initiation factor 2B. Although rare, with a reported prevalence of 1:80,000 (0.001%), it was considered as one of the most common leukodystrophies. However, the worldwide incidence and prevalence of this disease are not clear. In Bahrain, of 21 patients who were diagnosed with leukodystrophy, two patients were found to have VWM disease accounting for 9.5%. Vaccinations and infections were the trigger factors for this disease to manifest. Rapid neurological deterioration, loss of developmental milestones, and seizure disorders are the main presentations in both patients. Magnetic resonance imaging (MRI) showed the classical radiological changes of demyelination and leukodystrophy. Patient 1 had associated ulcerative colitis, a finding that was not reported before. Patient 1's condition progressed to a vegetative stage, while patient 2 passed away, reflecting the poor disease outcome. In patient 2, a novel homozygous missense mutation was found in the EIF2B3 gene (c.25G>A, p.Ala9Thr). In this report, we present in detail the prevalence of VWM disease among cases with leukodystrophy, patients' characteristics, clinical presentations, radiological findings, associated diseases, genetic results, and clinical outcomes in the main tertiary hospital in Bahrain between 1998 and 2024. Moreover, we conducted a thorough literature review on this rare condition.

摘要

消失性白质(VWM)病是一种常染色体隐性疾病,由编码真核起始因子2B亚基1-5的EIF2B基因突变引起。尽管罕见,报道的患病率为1:80,000(0.001%),但它曾被认为是最常见的脑白质营养不良症之一。然而,这种疾病在全球的发病率和患病率尚不清楚。在巴林,21例被诊断为脑白质营养不良症的患者中,有2例被发现患有VWM病,占9.5%。疫苗接种和感染是该疾病显现的触发因素。快速的神经功能恶化、发育里程碑丧失和癫痫障碍是这两名患者的主要表现。磁共振成像(MRI)显示了脱髓鞘和脑白质营养不良的典型影像学变化。患者1伴有溃疡性结肠炎,这一发现此前未见报道。患者1的病情进展至植物人状态,而患者2去世,反映出该疾病预后不佳。在患者2中,在EIF2B3基因中发现了一个新的纯合错义突变(c.25G>A,p.Ala9Thr)。在本报告中,我们详细介绍了1998年至2024年期间巴林主要三级医院中患有脑白质营养不良症的病例中VWM病的患病率、患者特征、临床表现、影像学检查结果、相关疾病、基因检测结果及临床结局。此外,我们对这种罕见疾病进行了全面的文献综述。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0f25/11645671/6d6d80999485/cureus-0016-00000073667-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0f25/11645671/687c08c21580/cureus-0016-00000073667-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0f25/11645671/6d6d80999485/cureus-0016-00000073667-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0f25/11645671/687c08c21580/cureus-0016-00000073667-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0f25/11645671/6d6d80999485/cureus-0016-00000073667-i02.jpg

相似文献

1
Vanishing White Matter Disease in Children: An Unusual Association, a Novel Mutation, and a Literature Review.儿童消失性白质病:一种不寻常的关联、一种新突变及文献综述
Cureus. 2024 Nov 14;16(11):e73667. doi: 10.7759/cureus.73667. eCollection 2024 Nov.
2
Mutations in the genes encoding eukaryotic translation initiation factor 2B in Japanese patients with vanishing white matter disease.日本儿童脑白质消失症患者中编码真核生物翻译起始因子2B的基因突变情况。
Brain Dev. 2015 Nov;37(10):960-6. doi: 10.1016/j.braindev.2015.03.003. Epub 2015 Apr 3.
3
Leukoencephalopathy with vanishing white matter disease: a case report study.伴脑白质消失的白质脑病:一项病例报告研究
Ann Med Surg (Lond). 2023 Jun 28;85(8):4087-4091. doi: 10.1097/MS9.0000000000001017. eCollection 2023 Aug.
4
Identification of a Missense Variant in the EIF2B3 Gene Causing Vanishing White Matter Disease with Antenatal-Onset but Mild Symptoms and Long-Term Survival.鉴定 EIF2B3 基因中的错义变异导致具有产前起病但症状轻微和长期生存的脑白质消融症。
J Mol Neurosci. 2021 Nov;71(11):2405-2414. doi: 10.1007/s12031-021-01810-0. Epub 2021 Mar 9.
5
Vanishing white matter disease with different faces.具有不同表现形式的脑白质消失症。
Childs Nerv Syst. 2020 Feb;36(2):353-361. doi: 10.1007/s00381-019-04334-6. Epub 2019 Aug 5.
6
Vanishing white matter disease in French-Canadian patients from Quebec.来自魁北克的法裔加拿大患者的脑白质消失症
Pediatr Neurol. 2014 Aug;51(2):225-32. doi: 10.1016/j.pediatrneurol.2014.05.006. Epub 2014 May 14.
7
Correlation Between Vanishing White Matter Disease and Novel Heterozygous EIF2B3 Variants Using Next-Generation Sequencing: A Case Report.使用下一代测序技术分析消失性白质脑病与新型杂合EIF2B3变体之间的相关性:一例报告
Ann Rehabil Med. 2019 Apr;43(2):234-238. doi: 10.5535/arm.2019.43.2.234. Epub 2019 Apr 30.
8
A Japanese girl with an early-infantile onset vanishing white matter disease resembling Cree leukoencephalopathy.一名患有类似克里白质脑病的早发性婴儿型消失性白质病的日本女孩。
Brain Dev. 2015 Jun;37(6):638-42. doi: 10.1016/j.braindev.2014.10.002. Epub 2014 Oct 27.
9
A rare case of adult-onset vanishing white matter leukoencephalopathy with movement disorder, expressing homozygous EIF2B3 and PRKN pathogenic variants.一例罕见的成年起病的伴运动障碍的消失性白质脑病,表达纯合子EIF2B3和PRKN致病变异。
BMC Neurol. 2025 Jan 4;25(1):7. doi: 10.1186/s12883-024-04018-y.
10
Identification of novel EIF2B mutations in Chinese patients with vanishing white matter disease.鉴定中国脑白质消融症患者中的新型 EIF2B 突变。
J Hum Genet. 2009 Feb;54(2):74-7. doi: 10.1038/jhg.2008.10. Epub 2009 Jan 16.

本文引用的文献

1
The Leukodystrophy Spectrum in Saudi Arabia: Epidemiological, Clinical, Radiological, and Genetic Data.沙特阿拉伯的脑白质营养不良谱系:流行病学、临床、放射学和遗传学数据。
Front Pediatr. 2021 May 13;9:633385. doi: 10.3389/fped.2021.633385. eCollection 2021.
2
Identification of a Missense Variant in the EIF2B3 Gene Causing Vanishing White Matter Disease with Antenatal-Onset but Mild Symptoms and Long-Term Survival.鉴定 EIF2B3 基因中的错义变异导致具有产前起病但症状轻微和长期生存的脑白质消融症。
J Mol Neurosci. 2021 Nov;71(11):2405-2414. doi: 10.1007/s12031-021-01810-0. Epub 2021 Mar 9.
3
Vanishing white matter: deregulated integrated stress response as therapy target.
消失的白质:失调的整合应激反应作为治疗靶点。
Ann Clin Transl Neurol. 2019 Aug;6(8):1407-1422. doi: 10.1002/acn3.50826. Epub 2019 Jul 18.
4
Vanishing white matter disease with different faces.具有不同表现形式的脑白质消失症。
Childs Nerv Syst. 2020 Feb;36(2):353-361. doi: 10.1007/s00381-019-04334-6. Epub 2019 Aug 5.
5
A Review of Infantile Vanishing White Matter Disease and A New Mutation.婴儿型消失性白质病综述及一种新突变
Acta Neurol Taiwan. 2017 Dec 15;26(4):167-176.
6
Natural History of Vanishing White Matter.脑白质消融症的自然病史。
Ann Neurol. 2018 Aug;84(2):274-288. doi: 10.1002/ana.25287. Epub 2018 Sep 6.
7
Similarities and differences between infantile and early childhood onset vanishing white matter disease.婴儿型和早发型进行性脑白质营养不良的异同。
J Neurol. 2018 Jun;265(6):1410-1418. doi: 10.1007/s00415-018-8851-6. Epub 2018 Apr 16.
8
Vanishing white matter disease in a spanish population.西班牙人群中的脑白质消失症
J Cent Nerv Syst Dis. 2014 Jul 13;6:59-68. doi: 10.4137/JCNSD.S13540. eCollection 2014.
9
Vanishing white matter in Saudi Arabia.沙特阿拉伯的脑白质消失症。
Neurosciences (Riyadh). 2008 Oct;13(4):433-6.
10
Vanishing white matter disease.脑白质消失症
Lancet Neurol. 2006 May;5(5):413-23. doi: 10.1016/S1474-4422(06)70440-9.