• 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

一种利用外周血单核细胞(PBMCs)新生蛋白质组来揭示脆性 X 蛋白组生物标志物的新策略。

A new strategy to uncover fragile X proteomic biomarkers using the nascent proteome of peripheral blood mononuclear cells (PBMCs).

机构信息

Department of Biochemistry and Functional Genomic, Faculty of Medicine and Health Sciences, Université de Sherbrooke and Centre de Recherche du CHUS, CIUSSS de l'Estrie-CHUS, Sherbrooke, QC, Canada.

出版信息

Sci Rep. 2021 Jul 26;11(1):15148. doi: 10.1038/s41598-021-94027-5.

DOI:10.1038/s41598-021-94027-5
PMID:34312401
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8313568/
Abstract

Fragile X syndrome (FXS) is the most prevalent inherited cause of intellectual disabilities and autism spectrum disorders. FXS result from the loss of expression of the FMRP protein, an RNA-binding protein that regulates the expression of key synaptic effectors. FXS is also characterized by a wide array of behavioural, cognitive and metabolic impairments. The severity and penetrance of those comorbidities are extremely variable, meaning that a considerable phenotypic heterogeneity is found among fragile X individuals. Unfortunately, clinicians currently have no tools at their disposal to assay a patient prognosis upon diagnosis. Since the absence of FMRP was repeatedly associated with an aberrant protein synthesis, we decided to study the nascent proteome in order to screen for potential proteomic biomarkers of FXS. We used a BONCAT (Biorthogonal Non-canonical Amino Acids Tagging) method coupled to label-free mass spectrometry to purify and quantify nascent proteins of peripheral blood mononuclear cells (PBMCs) from 7 fragile X male patients and 7 age-matched controls. The proteomic analysis identified several proteins which were either up or downregulated in PBMCs from FXS individuals. Eleven of those proteins were considered as potential biomarkers, of which 5 were further validated by Western blot. The gene ontology enrichment analysis highlighted molecular pathways that may contribute to FXS physiopathology. Our results suggest that the nascent proteome of PBMCs is well suited for the discovery of FXS biomarkers.

摘要

脆性 X 综合征 (FXS) 是最常见的遗传性智力障碍和自闭症谱系障碍的病因。FXS 是由于 FMRP 蛋白表达缺失引起的,FMRP 蛋白是一种 RNA 结合蛋白,可调节关键突触效应器的表达。FXS 还表现出广泛的行为、认知和代谢障碍。这些合并症的严重程度和外显率差异极大,这意味着脆性 X 个体之间存在相当大的表型异质性。不幸的是,临床医生目前没有工具可以在诊断时评估患者的预后。由于 FMRP 的缺失与异常的蛋白质合成反复相关,我们决定研究新生蛋白质组,以筛选 FXS 的潜在蛋白质组生物标志物。我们使用 BONCAT(双正交非规范氨基酸标记)方法结合无标记质谱法来纯化和定量来自 7 名脆性 X 男性患者和 7 名年龄匹配的对照者的外周血单核细胞 (PBMC) 中的新生蛋白质。蛋白质组学分析鉴定了 PBMC 中 FXS 个体中上调或下调的几种蛋白质。其中 11 种被认为是潜在的生物标志物,其中 5 种通过 Western blot 进一步验证。基因本体富集分析突出了可能有助于 FXS 病理生理学的分子途径。我们的结果表明,PBMC 的新生蛋白质组非常适合发现 FXS 生物标志物。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/6138f544cb06/41598_2021_94027_Fig6_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/1c60d4fad1aa/41598_2021_94027_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/f215680013e0/41598_2021_94027_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/6b6566d70852/41598_2021_94027_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/80eed72d2814/41598_2021_94027_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/847e2145b879/41598_2021_94027_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/6138f544cb06/41598_2021_94027_Fig6_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/1c60d4fad1aa/41598_2021_94027_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/f215680013e0/41598_2021_94027_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/6b6566d70852/41598_2021_94027_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/80eed72d2814/41598_2021_94027_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/847e2145b879/41598_2021_94027_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/418b/8313568/6138f544cb06/41598_2021_94027_Fig6_HTML.jpg

相似文献

1
A new strategy to uncover fragile X proteomic biomarkers using the nascent proteome of peripheral blood mononuclear cells (PBMCs).一种利用外周血单核细胞(PBMCs)新生蛋白质组来揭示脆性 X 蛋白组生物标志物的新策略。
Sci Rep. 2021 Jul 26;11(1):15148. doi: 10.1038/s41598-021-94027-5.
2
Rates of protein synthesis are reduced in peripheral blood mononuclear cells (PBMCs) from fragile X individuals.脆性 X 个体的外周血单核细胞 (PBMC) 中蛋白质合成率降低。
PLoS One. 2021 May 11;16(5):e0251367. doi: 10.1371/journal.pone.0251367. eCollection 2021.
3
Synaptic vesicle dynamic changes in a model of fragile X.脆性X模型中突触小泡的动态变化
Mol Autism. 2016 Mar 1;7:17. doi: 10.1186/s13229-016-0080-1. eCollection 2016.
4
Reduced serum levels of pro-inflammatory chemokines in fragile X syndrome.脆性X综合征患者血清促炎趋化因子水平降低。
BMC Neurol. 2020 Apr 15;20(1):138. doi: 10.1186/s12883-020-01715-2.
5
Reciprocal changes in DNA methylation and hydroxymethylation and a broad repressive epigenetic switch characterize FMR1 transcriptional silencing in fragile X syndrome.DNA甲基化和羟甲基化的相互变化以及广泛的抑制性表观遗传开关是脆性X综合征中FMR1转录沉默的特征。
Clin Epigenetics. 2016 Feb 5;8:15. doi: 10.1186/s13148-016-0181-x. eCollection 2016.
6
Epigenetic characterization of the FMR1 gene and aberrant neurodevelopment in human induced pluripotent stem cell models of fragile X syndrome.脆性 X 综合征患者诱导多能干细胞模型中 FMR1 基因的表观遗传学特征及神经发育异常。
PLoS One. 2011;6(10):e26203. doi: 10.1371/journal.pone.0026203. Epub 2011 Oct 12.
7
New insights of altered lipid profile in Fragile X Syndrome.脆性X综合征脂质谱改变的新见解。
PLoS One. 2017 Mar 23;12(3):e0174301. doi: 10.1371/journal.pone.0174301. eCollection 2017.
8
ICAM5 as a Novel Target for Treating Cognitive Impairment in Fragile X Syndrome.ICAM5 作为治疗脆性 X 综合征认知障碍的新靶点。
J Neurosci. 2020 Feb 5;40(6):1355-1365. doi: 10.1523/JNEUROSCI.2626-18.2019. Epub 2019 Dec 27.
9
Fmr1 deficiency promotes age-dependent alterations in the cortical synaptic proteome.Fmr1基因缺陷会促进皮质突触蛋白质组中与年龄相关的改变。
Proc Natl Acad Sci U S A. 2015 Aug 25;112(34):E4697-706. doi: 10.1073/pnas.1502258112. Epub 2015 Aug 11.
10
Molecular and cellular aspects of mental retardation in the Fragile X syndrome: from gene mutation/s to spine dysmorphogenesis.脆性 X 综合征智力迟钝的分子和细胞方面:从基因突变/到脊柱畸形发生。
Adv Exp Med Biol. 2012;970:517-51. doi: 10.1007/978-3-7091-0932-8_23.

引用本文的文献

1
Deciphering the physiopathology of neurodevelopmental disorders using brain organoids.利用脑类器官解读神经发育障碍的生理病理学
Brain. 2025 Jan 7;148(1):12-26. doi: 10.1093/brain/awae281.
2
Proteomics insights into fragile X syndrome: Unraveling molecular mechanisms and therapeutic avenues.蛋白质组学揭示脆性 X 综合征的奥秘:解析分子机制与治疗途径。
Neurobiol Dis. 2024 May;194:106486. doi: 10.1016/j.nbd.2024.106486. Epub 2024 Mar 26.
3
TIAR and FMRP shape pro-survival nascent proteome of leukemia cells in the bone marrow microenvironment.

本文引用的文献

1
An "Omic" Overview of Fragile X Syndrome.脆性X综合征的“组学”概述
Biology (Basel). 2021 May 13;10(5):433. doi: 10.3390/biology10050433.
2
Rates of protein synthesis are reduced in peripheral blood mononuclear cells (PBMCs) from fragile X individuals.脆性 X 个体的外周血单核细胞 (PBMC) 中蛋白质合成率降低。
PLoS One. 2021 May 11;16(5):e0251367. doi: 10.1371/journal.pone.0251367. eCollection 2021.
3
The molecular biology of FMRP: new insights into fragile X syndrome.脆性 X 综合征的 FMRP 分子生物学:新见解。
TIAR和FMRP塑造骨髓微环境中白血病细胞的促生存新生蛋白质组。
iScience. 2023 Mar 31;26(4):106543. doi: 10.1016/j.isci.2023.106543. eCollection 2023 Apr 21.
4
Potential of Multiscale Astrocyte Imaging for Revealing Mechanisms Underlying Neurodevelopmental Disorders.多尺度星形胶质细胞成像揭示神经发育障碍相关机制的潜力。
Int J Mol Sci. 2021 Sep 24;22(19):10312. doi: 10.3390/ijms221910312.
Nat Rev Neurosci. 2021 Apr;22(4):209-222. doi: 10.1038/s41583-021-00432-0. Epub 2021 Feb 19.
4
A Genotype-Phenotype Study of High-Resolution Nucleic Acid and Protein Analyses in Fragile X Patients with Neurobehavioral Assessments.一项针对伴有神经行为评估的脆性X综合征患者进行高分辨率核酸和蛋白质分析的基因型-表型研究。
Brain Sci. 2020 Sep 30;10(10):694. doi: 10.3390/brainsci10100694.
5
Optimization of Protocols for Detection of De Novo Protein Synthesis in Whole Blood Samples via Azide-Alkyne Cycloaddition.通过叠氮-炔基环加成反应优化全血样本中从头蛋白质合成检测的方案。
J Proteome Res. 2020 Sep 4;19(9):3856-3866. doi: 10.1021/acs.jproteome.0c00299. Epub 2020 Aug 20.
6
Blood-Based Biomarkers Predictive of Metformin Target Engagement in Fragile X Syndrome.基于血液的生物标志物可预测二甲双胍在脆性X综合征中的靶点参与情况。
Brain Sci. 2020 Jun 10;10(6):361. doi: 10.3390/brainsci10060361.
7
Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome.一项针对脆性 X 综合征青少年的洛伐他汀联合开放式父母实施语言干预的对照试验。
J Neurodev Disord. 2020 Apr 22;12(1):12. doi: 10.1186/s11689-020-09315-4.
8
Association between IQ and FMR1 protein (FMRP) across the spectrum of CGG repeat expansions.在 CGG 重复扩展的整个范围内,智商与 FMR1 蛋白(FMRP)之间的关联。
PLoS One. 2019 Dec 31;14(12):e0226811. doi: 10.1371/journal.pone.0226811. eCollection 2019.
9
Ser-Phosphorylation of PCSK9 (Proprotein Convertase Subtilisin-Kexin 9) by Fam20C (Family With Sequence Similarity 20, Member C) Kinase Enhances Its Ability to Degrade the LDLR (Low-Density Lipoprotein Receptor).丝氨酸磷酸化的前蛋白转化酶枯草溶菌素 9(Proprotein Convertase Subtilisin-Kexin 9)通过 Fam20C(家族与序列相似性 20,成员 C)激酶增强其降解 LDLR(低密度脂蛋白受体)的能力。
Arterioscler Thromb Vasc Biol. 2019 Oct;39(10):1996-2013. doi: 10.1161/ATVBAHA.119.313247. Epub 2019 Sep 5.
10
Molecular Biomarkers in Fragile X Syndrome.脆性X综合征中的分子生物标志物
Brain Sci. 2019 Apr 27;9(5):96. doi: 10.3390/brainsci9050096.