• 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

线粒体 T10432C 突变对 Cika 牛的影响:Leber 遗传性视神经病变的“潜在”模型。

The Consequences of Mitochondrial T10432C Mutation in Cika Cattle: A "Potential" Model for Leber's Hereditary Optic Neuropathy.

机构信息

Department of Pathology, Croatian Veterinary Institute, 10000 Zagreb, Croatia.

Department of Animal Science, Faculty of Agriculture, University of Zagreb, 10000 Zagreb, Croatia.

出版信息

Int J Mol Sci. 2022 Jun 6;23(11):6335. doi: 10.3390/ijms23116335.

DOI:10.3390/ijms23116335
PMID:35683014
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9181260/
Abstract

While mitogenome mutations leading to pathological manifestations are rare, more than 200 such mutations have been described in humans. In contrast, pathogenic mitogenome mutations are rare in domestic animals and have not been described at all in cattle. In the small local Slovenian cattle breed Cika, we identified (next-generation sequencing) two cows with the T10432C mitogenome mutation in the ND4L gene, which corresponds to the human T10663C mutation known to cause Leber's hereditary optic neuropathy (LHON). Pedigree analysis revealed that the cows in which the mutation was identified belong to two different maternal lineages with 217 individual cows born between 1997 and 2020. The identified mutation and its maternal inheritance were confirmed by Sanger sequencing across multiple generations, whereas no single analysis revealed evidence of heteroplasmy. A closer clinical examination of one cow with the T10432C mutation revealed exophthalmos, whereas histopathological examination revealed retinal ablations, subretinal oedema, and haemorrhage. The results of these analyses confirm the presence of mitochondrial mutation T10432C with homoplasmic maternal inheritance as well as clinical and histopathological signs similar to LHON in humans. Live animals with the mutation could be used as a suitable animal model that can improve our understanding of the pathogenesis of LHON and other mitochondriopathies.

摘要

虽然导致病理性表现的线粒体基因突变很少见,但已经在人类中描述了超过 200 种这样的突变。相比之下,致病性线粒体基因突变在家畜中很少见,在牛中根本没有描述。在斯洛文尼亚的小型本地 Cika 牛品种中,我们通过(下一代测序)在 ND4L 基因中发现了两只 T10432C 线粒体突变的奶牛,这与已知导致 Leber 遗传性视神经病变(LHON)的人类 T10663C 突变相对应。系谱分析显示,发现突变的奶牛属于两个不同的母系,1997 年至 2020 年间出生了 217 头奶牛。通过对多个世代进行 Sanger 测序,确认了所鉴定的突变及其母系遗传,而没有单一分析显示异质性的证据。对携带 T10432C 突变的一只奶牛进行更密切的临床检查发现了眼球突出,而组织病理学检查则发现视网膜消融、视网膜下水肿和出血。这些分析的结果证实了具有纯合母系遗传的线粒体突变 T10432C 的存在,以及与人类 LHON 相似的临床和组织病理学迹象。携带该突变的活体动物可用作合适的动物模型,有助于我们更好地理解 LHON 和其他线粒体疾病的发病机制。

相似文献

1
The Consequences of Mitochondrial T10432C Mutation in Cika Cattle: A "Potential" Model for Leber's Hereditary Optic Neuropathy.线粒体 T10432C 突变对 Cika 牛的影响:Leber 遗传性视神经病变的“潜在”模型。
Int J Mol Sci. 2022 Jun 6;23(11):6335. doi: 10.3390/ijms23116335.
2
Peculiar combinations of individually non-pathogenic missense mitochondrial DNA variants cause low penetrance Leber's hereditary optic neuropathy.独特组合的个体非致病性错义线粒体 DNA 变异导致低外显率的莱伯遗传性视神经病变。
PLoS Genet. 2018 Feb 14;14(2):e1007210. doi: 10.1371/journal.pgen.1007210. eCollection 2018 Feb.
3
In silico model of mtDNA mutations effect on secondary and 3D structure of mitochondrial rRNA and tRNA in Leber's hereditary optic neuropathy.利伯遗传性视神经病变中线粒体 rRNA 和 tRNA 二级和三维结构的 mtDNA 突变效应的计算机模型。
Exp Eye Res. 2020 Dec;201:108277. doi: 10.1016/j.exer.2020.108277. Epub 2020 Sep 28.
4
ND4L gene concurrent 10609T>C and 10663T>C mutations are associated with Leber's hereditary optic neuropathy in a large pedigree from Kuwait.ND4L 基因同时存在 10609T>C 和 10663T>C 突变与科威特一个大家系中的莱伯遗传性视神经病变有关。
Br J Ophthalmol. 2014 Jun;98(6):826-31. doi: 10.1136/bjophthalmol-2013-304140. Epub 2014 Feb 25.
5
Frequency and spectrum of mitochondrial ND6 mutations in 1218 Han Chinese subjects with Leber's hereditary optic neuropathy.1218 例汉族莱伯遗传性视神经病变患者中线粒体 ND6 突变的频率和谱。
Invest Ophthalmol Vis Sci. 2014 Mar 6;55(3):1321-31. doi: 10.1167/iovs.13-13011.
6
Prevalence of Mitochondrial ND4 Mutations in 1281 Han Chinese Subjects With Leber's Hereditary Optic Neuropathy.1281例中国汉族Leber遗传性视神经病变患者线粒体ND4基因突变的患病率
Invest Ophthalmol Vis Sci. 2015 Jul;56(8):4778-88. doi: 10.1167/iovs.14-16158.
7
m.3635G>A mutation as a cause of Leber hereditary optic neuropathy.m.3635G>A 突变是 Leber 遗传性视神经病变的原因。
J Clin Pathol. 2014 Jul;67(7):639-41. doi: 10.1136/jclinpath-2014-202192. Epub 2014 Apr 19.
8
[Past, present, and future in Leber's hereditary optic neuropathy].[莱伯遗传性视神经病变的过去、现在与未来]
Nippon Ganka Gakkai Zasshi. 2001 Dec;105(12):809-27.
9
A simple oligonucleotide biochip capable of rapidly detecting known mitochondrial DNA mutations in Chinese patients with Leber's hereditary optic neuropathy (LHON).一种简单的寡核苷酸生物芯片,能够快速检测中国莱伯遗传性视神经病变(LHON)患者已知的线粒体 DNA 突变。
Dis Markers. 2011;30(4):181-90. doi: 10.3233/DMA-2011-0767.
10
[A pedigree of Leber's hereditary optic neuropathy with mtDNA 14484 mutation].[伴有线粒体DNA 14484突变的Leber遗传性视神经病变家系]
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2008 Oct;25(5):531-3.

本文引用的文献

1
Large-scale mitogenome sequencing reveals consecutive expansions of domestic taurine cattle and supports sporadic aurochs introgression.大规模线粒体基因组测序揭示了家养黄牛的连续扩张并支持原牛的零星基因渗入。
Evol Appl. 2021 Nov 27;15(4):663-678. doi: 10.1111/eva.13315. eCollection 2022 Apr.
2
Mitochondrial Retinopathies.线粒体视神经病。
Int J Mol Sci. 2021 Dec 25;23(1):210. doi: 10.3390/ijms23010210.
3
Estimation of Genetic Parameters of Type Traits in First Parity Cows of the Autochthonous Cika Cattle in Slovenia.斯洛文尼亚本土契卡牛头胎母牛类型性状遗传参数的估计
Front Genet. 2021 Nov 22;12:724058. doi: 10.3389/fgene.2021.724058. eCollection 2021.
4
Hierarchical Modelling of Haplotype Effects on a Phylogeny.单倍型对系统发育影响的层次建模
Front Genet. 2021 Jan 15;11:531218. doi: 10.3389/fgene.2020.531218. eCollection 2020.
5
Polygenic inheritance, GWAS, polygenic risk scores, and the search for functional variants.多基因遗传、GWAS、多基因风险评分与功能变异的寻找。
Proc Natl Acad Sci U S A. 2020 Aug 11;117(32):18924-18933. doi: 10.1073/pnas.2005634117. Epub 2020 Aug 4.
6
Mitochondrial Disorder: Kearns-Sayre Syndrome.线粒体疾病:肌阵挛性癫痫伴破碎红纤维(Kearns-Sayre 综合征)。
Adv Exp Med Biol. 2018;1085:161-162. doi: 10.1007/978-3-319-95046-4_30.
7
Genome-wide association analyses identify 44 risk variants and refine the genetic architecture of major depression.全基因组关联分析确定了 44 个风险变异,并完善了重度抑郁症的遗传结构。
Nat Genet. 2018 May;50(5):668-681. doi: 10.1038/s41588-018-0090-3. Epub 2018 Apr 26.
8
MaGelLAn 1.0: a software to facilitate quantitative and population genetic analysis of maternal inheritance by combination of molecular and pedigree information.麦哲伦1.0:一款通过整合分子和谱系信息来促进母系遗传定量和群体遗传分析的软件。
Genet Sel Evol. 2016 Sep 10;48(1):65. doi: 10.1186/s12711-016-0242-9.
9
Recovery of native genetic background in admixed populations using haplotypes, phenotypes, and pedigree information--using Cika cattle as a case breed.利用单倍型、表型和系谱信息恢复混合群体中的原生遗传背景——以契卡牛为例。
PLoS One. 2015 Apr 29;10(4):e0123253. doi: 10.1371/journal.pone.0123253. eCollection 2015.
10
Mitochondrial diseases.线粒体疾病
J Epilepsy Res. 2012 Mar 30;2(1):1-4. doi: 10.14581/jer.12001. eCollection 2012 Mar.