• 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

高-莫瓦特综合征:一种与肾功能损害相关的早发性进行性脑病,伴有难治性癫痫。两例新病例及文献复习。

Galloway-Mowat syndrome: an early-onset progressive encephalopathy with intractable epilepsy associated to renal impairment. Two novel cases and review of literature.

机构信息

Muscular and Neurodegenerative Disease Unit, Institute G. Gaslini, Genova, Italy.

出版信息

Seizure. 2010 Mar;19(2):132-5. doi: 10.1016/j.seizure.2009.12.002. Epub 2010 Jan 18.

DOI:10.1016/j.seizure.2009.12.002
PMID:20083416
Abstract

Galloway-Mowat Syndrome (GMS) is an autosomal recessively inherited condition which manifests with severe encephalopathy, featuring microcephaly, developmental delay, and early-onset intractable epilepsy. Patients typically show also renal involvement from the onset. We report two siblings with GMS presenting with early-onset, intractable epilepsy and neurological deterioration, later followed by renal impairment. In both patients intractable epilepsy started during the first months of life and included a combination of spasms, focal and myoclonic/atonic seizures, along with psychomotor retardation and dysmorphic features. One of the patient died from fulminating renal failure at age 6 years. The other patient developed only isolated proteinuria from the age 3 years. Our cases differ from 'classic' GMS, as manifested the clinical and laboratory features of renal involvement only some years later the onset of epilepsy and neurological symptoms. Therefore, the diagnosis of GMS should be considered in infants with intractable epilepsy, encephalopathy, and multiple neurological deficits, also in absence of renal manifestations. The literature data about the electroclinical features of epilepsy in GMS are also reviewed.

摘要

Galloway-Mowat 综合征(GMS)是一种常染色体隐性遗传疾病,表现为严重的脑病,特征为小头畸形、发育迟缓以及早发性难治性癫痫。患者通常从发病开始就有肾脏受累。我们报告了两例 GMS 同胞,表现为早发性、难治性癫痫和神经功能恶化,随后出现肾功能损害。在这两例患者中,难治性癫痫均在生命的头几个月开始,包括痉挛、局灶性和肌阵挛/失张力发作,伴有精神运动发育迟缓和发育异常。其中一名患者在 6 岁时死于暴发性肾衰竭。另一名患者从 3 岁起仅出现孤立性蛋白尿。我们的病例与“经典”GMS 不同,因为仅在癫痫和神经症状发作几年后才表现出肾脏受累的临床和实验室特征。因此,对于有难治性癫痫、脑病和多种神经功能缺损的婴儿,即使无肾脏表现,也应考虑 GMS 的诊断。还回顾了 GMS 癫痫的电临床特征的文献数据。

相似文献

1
Galloway-Mowat syndrome: an early-onset progressive encephalopathy with intractable epilepsy associated to renal impairment. Two novel cases and review of literature.高-莫瓦特综合征:一种与肾功能损害相关的早发性进行性脑病,伴有难治性癫痫。两例新病例及文献复习。
Seizure. 2010 Mar;19(2):132-5. doi: 10.1016/j.seizure.2009.12.002. Epub 2010 Jan 18.
2
Late-onset nephrotic syndrome and severe cerebellar atrophy in Galloway-Mowat syndrome.加洛韦-莫瓦特综合征中的迟发性肾病综合征和严重小脑萎缩。
Neuropediatrics. 2005 Oct;36(5):332-5. doi: 10.1055/s-2005-872842.
3
Collapsing glomerulopathy in Galloway-Mowat syndrome: a case report and review of the literature.加洛韦-莫瓦特综合征中的塌陷性肾小球病:一例报告并文献复习
Pathol Res Pract. 2008;204(6):401-6. doi: 10.1016/j.prp.2007.12.007. Epub 2008 Feb 13.
4
Late-onset nephrotic syndrome in galloway-mowat syndrome: a case report.加洛韦-莫瓦特综合征中的迟发性肾病综合征:一例报告。
Saudi J Kidney Dis Transpl. 1999 Apr-Jun;10(2):171-4.
5
Severe idiopathic generalized epilepsy of infancy with generalized tonic-clonic seizures.伴有全身强直阵挛发作的婴儿严重特发性全身性癫痫
Neuropediatrics. 1998 Oct;29(5):229-38. doi: 10.1055/s-2007-973567.
6
Epilepsy and mental retardation limited to females: an under-recognized disorder.仅见于女性的癫痫和智力发育迟缓:一种未被充分认识的疾病。
Brain. 2008 Apr;131(Pt 4):918-27. doi: 10.1093/brain/awm338. Epub 2008 Jan 29.
7
Ring 14 chromosome presenting as early-onset isolated partial epilepsy.14号环状染色体表现为早发性孤立性部分性癫痫。
Dev Med Child Neurol. 2009 Nov;51(11):917-22. doi: 10.1111/j.1469-8749.2009.03292.x. Epub 2009 Mar 31.
8
Hypothalamic hamartoma and epilepsy in children: illustrative cases of possible evolutions.儿童下丘脑错构瘤与癫痫:可能演变的实例
Epileptic Disord. 2003 Dec;5(4):187-99.
9
Neuropathological homology in true Galloway-Mowat syndrome.真性加洛韦-莫瓦特综合征的神经病理学同源性。
J Child Neurol. 2011 Apr;26(4):510-7. doi: 10.1177/0883073810383982. Epub 2011 Jan 13.
10
Galloway-Mowat syndrome.加洛韦-莫瓦特综合征
J Coll Physicians Surg Pak. 2008 Aug;18(8):520-1.

引用本文的文献

1
Refining the Phenotypic and Genotypic Spectrum of -Related Galloway-Mowat Syndrome: A Case Series and Systematic Review.细化与加洛韦-莫瓦特综合征相关的表型和基因型谱:病例系列及系统评价
Neurol Genet. 2025 Jul 11;11(4):e200280. doi: 10.1212/NXG.0000000000200280. eCollection 2025 Aug.
2
Genetics and phenotypic heterogeneity of Galloway-Mowat syndrome.加洛韦-莫瓦特综合征的遗传学与表型异质性
Cell Commun Signal. 2025 Jun 18;23(1):289. doi: 10.1186/s12964-025-02307-8.
3
Whole-exome sequencing revealed a novel homozygous missense variant in OSGEP gene: a case report of Galloway-Mowat syndrome in Iran.
全外显子组测序揭示了 OSGEP 基因中的一个新型纯合错义变异:伊朗一例 Galloway-Mowat 综合征。
CEN Case Rep. 2023 Nov;12(4):374-377. doi: 10.1007/s13730-023-00775-w. Epub 2023 Mar 1.
4
Disruption of pathways regulated by Integrator complex in Galloway-Mowat syndrome due to WDR73 mutations.WDR73 突变导致 Galloway-Mowat 综合征中整合复合物调控通路的破坏。
Sci Rep. 2021 Mar 8;11(1):5388. doi: 10.1038/s41598-021-84472-7.
5
Movement Disorders and Renal Diseases.运动障碍与肾脏疾病
Mov Disord Clin Pract. 2020 Aug 10;7(7):763-779. doi: 10.1002/mdc3.13005. eCollection 2020 Oct.
6
WD40-Repeat Proteins in Ciliopathies and Congenital Disorders of Endocrine System.WD40 重复蛋白在纤毛病和先天性内分泌系统疾病中的作用。
Endocrinol Metab (Seoul). 2020 Sep;35(3):494-506. doi: 10.3803/EnM.2020.302. Epub 2020 Sep 8.
7
Extending the ophthalmological phenotype of Galloway-Mowat syndrome with distinct retinal dysfunction: a report and review of ocular findings.扩展伴有独特视网膜功能障碍的加洛韦-莫瓦特综合征的眼科表型:一份报告及眼部检查结果综述
BMC Ophthalmol. 2018 Jun 22;18(1):147. doi: 10.1186/s12886-018-0820-4.
8
Collapsing Glomerulopathy in a Child with Galloway-Mowat Syndrome.一名患有加洛韦-莫瓦特综合征儿童的塌陷性肾小球病
Case Rep Nephrol. 2016;2016:4386291. doi: 10.1155/2016/4386291. Epub 2016 Jun 14.
9
Extending the mutation spectrum for Galloway-Mowat syndrome to include homozygous missense mutations in the WDR73 gene.将加洛韦-莫瓦特综合征的突变谱扩展至包括WDR73基因中的纯合错义突变。
Am J Med Genet A. 2016 Apr;170A(4):992-8. doi: 10.1002/ajmg.a.37533. Epub 2016 Jan 5.
10
Recessive nephrocerebellar syndrome on the Galloway-Mowat syndrome spectrum is caused by homozygous protein-truncating mutations of WDR73.加洛韦-莫瓦特综合征谱系中的隐性肾小脑综合征由WDR73的纯合蛋白截短突变引起。
Brain. 2015 Aug;138(Pt 8):2173-90. doi: 10.1093/brain/awv153. Epub 2015 Jun 11.