• 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

一名埃及患者因戊二酰辅酶A脱氢酶基因发生新型移码突变而出现严重神经表现。

Severe neurological manifestations in an Egyptian patient with a novel frameshift mutation in the Glutaryl-CoA dehydrogenase gene.

作者信息

Moseilhy Ahmed, Hassan Magdy M, El Abd Heba S A, Mohammad Shaimaa A, El Bekay Rajaa, Abdel-Motal Ussama M, Ouhtit Allal, Zaki Osama K, Zayed Hatem

机构信息

Medical Genetics Unit, Pediatric Department, Faculty of Medicine, Ain Shams University Hospital, Cairo, 11665, Egypt.

Department of Biochemistry, Faculty of Science, Ain Shams University, Cairo, Egypt.

出版信息

Metab Brain Dis. 2017 Feb;32(1):35-40. doi: 10.1007/s11011-016-9879-x. Epub 2016 Aug 1.

DOI:10.1007/s11011-016-9879-x
PMID:27476540
Abstract

To characterize an Egyptian patient with glutaric acidemia type I (GA I) and to identify the causative mutation(s) that may be responsible for the disease phenotype. MRI was performed on the patient using the 1.5 T magnet, biochemical analysis was carried out using gas chromatography/mass spectrometry on the patient's dried blood spot, and the patient's organic acids were measured in dried blood and a urine sample using MS/MS and GC/MS, respectively. Total RNA was isolated from the patient's peripheral blood, and the synthesized cDNA was bi-directionally sequenced. The patient exhibited clinical features and MRI findings compatible with a diagnosis of GA I. The abnormal elevation of organic acids in the urine supported the presence of glutaryl-CoA dehydrogenase deficiency. Gene sequencing revealed a novel homozygous frameshift mutation, c.644_645insCTCG; p.(Pro217Leufs*14), in exon 8 of the GCDH gene. The present study revealed a novel frameshift mutation responsible for a severe GA I phenotype in an Egyptian patient. This novel mutation will ultimately contribute to a better understanding of the molecular pathology of the disease and shed light on the intricacies of the genotype-phenotype correlation of GA I disease.

摘要

为了对一名患有I型戊二酸血症(GA I)的埃及患者进行特征描述,并确定可能导致该疾病表型的致病突变。使用1.5 T磁体对患者进行了MRI检查,使用气相色谱/质谱法对患者的干血斑进行了生化分析,分别使用串联质谱法(MS/MS)和气相色谱/质谱法(GC/MS)对患者干血和尿液样本中的有机酸进行了测量。从患者外周血中分离出总RNA,并对合成的cDNA进行双向测序。该患者表现出与GA I诊断相符的临床特征和MRI结果。尿液中有机酸的异常升高支持戊二酰辅酶A脱氢酶缺乏的存在。基因测序揭示了GCDH基因第8外显子中的一个新的纯合移码突变,c.644_645insCTCG;p.(Pro217Leufs*14)。本研究揭示了一个导致埃及患者严重GA I表型的新的移码突变。这一新突变最终将有助于更好地理解该疾病的分子病理学,并阐明GA I疾病基因型-表型相关性的复杂性。

相似文献

1
Severe neurological manifestations in an Egyptian patient with a novel frameshift mutation in the Glutaryl-CoA dehydrogenase gene.一名埃及患者因戊二酰辅酶A脱氢酶基因发生新型移码突变而出现严重神经表现。
Metab Brain Dis. 2017 Feb;32(1):35-40. doi: 10.1007/s11011-016-9879-x. Epub 2016 Aug 1.
2
Genotype-phenotype correlation in 18 Egyptian patients with glutaric acidemia type I.18 例埃及戊二酸血症 I 型患者的基因型-表型相关性研究。
Metab Brain Dis. 2017 Oct;32(5):1417-1426. doi: 10.1007/s11011-017-0006-4. Epub 2017 Apr 7.
3
Clinical, biochemical, neuroradiological and molecular characterization of Egyptian patients with glutaric acidemia type 1.对 1 型戊二酸血症埃及患者的临床、生化、神经放射学和分子特征进行描述。
Metab Brain Dis. 2019 Aug;34(4):1231-1241. doi: 10.1007/s11011-019-00422-3. Epub 2019 May 6.
4
A Korean patient with glutaric aciduria type 1 with a novel mutation in the glutaryl CoA dehydrogenase gene.一名患有1型戊二酸尿症的韩国患者,其戊二酰辅酶A脱氢酶基因存在新的突变。
Ann Clin Lab Sci. 2014 Spring;44(2):213-6.
5
[Clinical investigation and genetic analysis of a Chinese family with glutaric acidemia type I].[一个中国I型戊二酸血症家系的临床调查与基因分析]
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2014 Oct;31(5):608-11. doi: 10.3760/cma.j.issn.1003-9406.2014.01.015.
6
Molecular genetic study of glutaric aciduria, type I: Identification of a novel mutation.I 型戊二酸血症的分子遗传学研究:一种新突变的鉴定。
J Cell Biochem. 2019 Mar;120(3):3367-3372. doi: 10.1002/jcb.27607. Epub 2018 Sep 11.
7
The M405V allele of the glutaryl-CoA dehydrogenase gene is an important marker for glutaric aciduria type I (GA-I) low excretors.戊二酰辅酶A脱氢酶基因的M405V等位基因是I型戊二酸血症(GA-I)低排泄者的重要标志物。
Mol Genet Metab. 2016 Sep;119(1-2):50-6. doi: 10.1016/j.ymgme.2016.06.012. Epub 2016 Jul 1.
8
[Mutation analysis of GCDH gene in four patients with glutaric academia type I].[4例Ⅰ型戊二酸血症患者GCDH基因的突变分析]
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2015 Apr;32(2):187-91. doi: 10.3760/cma.j.issn.1003-9406.2015.02.008.
9
Spectrum of mutations in Glutaryl-CoA dehydrogenase gene in glutaric aciduria type I--Study from South India.I型戊二酸血症中戊二酰辅酶A脱氢酶基因突变谱——来自印度南部的研究
Brain Dev. 2016 Jan;38(1):54-60. doi: 10.1016/j.braindev.2015.05.013. Epub 2015 Jun 10.
10
[Clinical phenotype and novel mutation in one of twins with glutaric acidemia type I].[一对I型戊二酸血症双胞胎之一的临床表型及新突变]
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2019 Jun 10;36(6):602-605. doi: 10.3760/cma.j.issn.1003-9406.2019.06.018.

引用本文的文献

1
Clinical, biochemical, neuroradiological and molecular characterization of Egyptian patients with glutaric acidemia type 1.对 1 型戊二酸血症埃及患者的临床、生化、神经放射学和分子特征进行描述。
Metab Brain Dis. 2019 Aug;34(4):1231-1241. doi: 10.1007/s11011-019-00422-3. Epub 2019 May 6.
2
Computational modelling approaches as a potential platform to understand the molecular genetics association between Parkinson's and Gaucher diseases.计算建模方法作为一种潜在的平台,用于了解帕金森病和戈谢病之间的分子遗传学关联。
Metab Brain Dis. 2018 Dec;33(6):1835-1847. doi: 10.1007/s11011-018-0286-3. Epub 2018 Jul 6.
3
Genotype-phenotype correlation in 18 Egyptian patients with glutaric acidemia type I.

本文引用的文献

1
Glutaric aciduria type 1: neuroimaging features with clinical correlation.1型戊二酸血症:神经影像学特征与临床相关性
Pediatr Radiol. 2015 Oct;45(11):1696-705. doi: 10.1007/s00247-015-3395-8. Epub 2015 Jun 26.
2
Glutaric Acidemia Type 1-Clinico-Molecular Profile and Novel Mutations in GCDH Gene in Indian Patients.1型戊二酸血症——印度患者的临床分子特征及GCDH基因新突变
JIMD Rep. 2015;21:45-55. doi: 10.1007/8904_2014_377. Epub 2015 Mar 12.
3
Clinical and molecular investigation of 19 Japanese cases of glutaric acidemia type 1.
18 例埃及戊二酸血症 I 型患者的基因型-表型相关性研究。
Metab Brain Dis. 2017 Oct;32(5):1417-1426. doi: 10.1007/s11011-017-0006-4. Epub 2017 Apr 7.
19 例日本 1 型戊二酸血症的临床和分子研究。
Mol Genet Metab. 2011 Mar;102(3):343-8. doi: 10.1016/j.ymgme.2010.11.159. Epub 2010 Nov 25.
4
Use of guidelines improves the neurological outcome in glutaric aciduria type I.指南的使用改善了Ⅰ型戊二酸血症的神经学预后。
Ann Neurol. 2010 Nov;68(5):743-52. doi: 10.1002/ana.22095.
5
Consanguinity and reproductive health among Arabs.阿拉伯人群中的近亲结婚与生殖健康
Reprod Health. 2009 Oct 8;6:17. doi: 10.1186/1742-4755-6-17.
6
Glutaric aciduria type I: outcome following detection by newborn screening.I型戊二酸血症:新生儿筛查检测后的结局
J Inherit Metab Dis. 2008 Aug;31(4):503-7. doi: 10.1007/s10545-008-0912-z. Epub 2008 Aug 9.
7
Newborn screening for glutaric aciduria type I in Victoria: treatment and outcome.维多利亚州新生儿戊二酸血症I型筛查:治疗与结果
Mol Genet Metab. 2008 Jul;94(3):287-91. doi: 10.1016/j.ymgme.2008.03.005. Epub 2008 Apr 14.
8
Decline of acute encephalopathic crises in children with glutaryl-CoA dehydrogenase deficiency identified by newborn screening in Germany.在德国通过新生儿筛查确诊的戊二酰辅酶A脱氢酶缺乏症儿童急性脑病危机的减少情况。
Pediatr Res. 2007 Sep;62(3):357-63. doi: 10.1203/PDR.0b013e318137a124.
9
Guideline for the diagnosis and management of glutaryl-CoA dehydrogenase deficiency (glutaric aciduria type I).戊二酰辅酶A脱氢酶缺乏症(I型戊二酸血症)诊断与管理指南
J Inherit Metab Dis. 2007 Feb;30(1):5-22. doi: 10.1007/s10545-006-0451-4. Epub 2007 Jan 3.
10
Natural history, outcome, and treatment efficacy in children and adults with glutaryl-CoA dehydrogenase deficiency.戊二酰辅酶A脱氢酶缺乏症儿童和成人的自然病史、预后及治疗效果
Pediatr Res. 2006 Jun;59(6):840-7. doi: 10.1203/01.pdr.0000219387.79887.86. Epub 2006 Apr 26.