• 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

推进脊髓和延髓肌肉萎缩症治疗的流行病学和遗传学方法:关注加拿大西部原住民中的患病率。

Advancing Epidemiology and Genetic Approaches for the Treatment of Spinal and Bulbar Muscular Atrophy: Focus on Prevalence in the Indigenous Population of Western Canada.

机构信息

Department of Medical Genetics, Faculty of Medicine and Dentistry, University of Alberta, Edmonton, AB T6G 2R3, Canada.

Department of Biological Sciences, Faculty of Science, University of Alberta, Edmonton, AB T6G 2R3, Canada.

出版信息

Genes (Basel). 2023 Aug 17;14(8):1634. doi: 10.3390/genes14081634.

DOI:10.3390/genes14081634
PMID:37628685
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10454234/
Abstract

Spinal and bulbar muscular atrophy (SBMA), also known as Kennedy's disease, is a debilitating neuromuscular disease characterized by progressive muscular weakness and neuronal degeneration, affecting 1-2 individuals per 100,000 globally. While SBMA is relatively rare, recent studies have shown a significantly higher prevalence of the disease among the indigenous population of Western Canada compared to the general population. The disease is caused by a pathogenic expansion of polyglutamine residues in the androgen receptor protein, which acts as a key transcriptional regulator for numerous genes. SBMA has no cure, and current treatments are primarily supportive and focused on symptom management. Recently, a form of precision medicine known as antisense therapy has gained traction as a promising therapeutic option for numerous neuromuscular diseases. Antisense therapy uses small synthetic oligonucleotides to confer therapeutic benefit by acting on pathogenic mRNA molecules, serving to either degrade pathogenic mRNA transcripts or helping to modulate splicing. Recent studies have explored the suitability of antisense therapy for the treatment of SBMA, primarily focused on gene therapy and antisense-mediated mRNA knockdown approaches. Advancements in understanding the pathogenesis of SBMA and the development of targeted therapies offer hope for improved quality of life for individuals affected by this debilitating condition. Continued research is essential to optimize these genetic approaches, ensuring their safety and efficacy.

摘要

脊髓延髓肌萎缩症(SBMA),又称肯尼迪病,是一种进行性肌肉无力和神经元退化的衰弱性神经肌肉疾病,全球每 10 万人中有 1-2 人受其影响。尽管 SBMA 相对罕见,但最近的研究表明,与一般人群相比,加拿大西部的原住民群体中该病的发病率明显更高。该病是由雄激素受体蛋白中多聚谷氨酰胺残基的病理性扩增引起的,该蛋白是许多基因的关键转录调节剂。SBMA 目前尚无治愈方法,现有的治疗方法主要是支持性的,侧重于症状管理。最近,一种称为反义疗法的精准医学形式作为许多神经肌肉疾病的有前途的治疗选择引起了关注。反义疗法使用小的合成寡核苷酸通过作用于致病的 mRNA 分子来发挥治疗作用,从而降解致病的 mRNA 转录物或有助于调节剪接。最近的研究探讨了反义疗法治疗 SBMA 的适用性,主要集中在基因治疗和反义介导的 mRNA 敲低方法上。对 SBMA 发病机制的深入了解和靶向治疗的发展为改善受这种衰弱性疾病影响的个体的生活质量带来了希望。持续的研究对于优化这些遗传方法至关重要,以确保其安全性和有效性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee16/10454234/5d78f812454e/genes-14-01634-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee16/10454234/de6ef9a3e686/genes-14-01634-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee16/10454234/5d78f812454e/genes-14-01634-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee16/10454234/de6ef9a3e686/genes-14-01634-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee16/10454234/5d78f812454e/genes-14-01634-g002.jpg

相似文献

1
Advancing Epidemiology and Genetic Approaches for the Treatment of Spinal and Bulbar Muscular Atrophy: Focus on Prevalence in the Indigenous Population of Western Canada.推进脊髓和延髓肌肉萎缩症治疗的流行病学和遗传学方法:关注加拿大西部原住民中的患病率。
Genes (Basel). 2023 Aug 17;14(8):1634. doi: 10.3390/genes14081634.
2
Deterioration of muscle force and contractile characteristics are early pathological events in spinal and bulbar muscular atrophy mice.肌肉力量和收缩特性的恶化是脊髓性肌萎缩症和延髓性肌肉萎缩症小鼠的早期病理事件。
Dis Model Mech. 2020 May 26;13(5):dmm042424. doi: 10.1242/dmm.042424.
3
Kennedy's disease (spinal and bulbar muscular atrophy): a clinically oriented review of a rare disease.肯尼迪病(脊髓延髓肌萎缩症):一种罕见疾病的临床综述。
J Neurol. 2019 Mar;266(3):565-573. doi: 10.1007/s00415-018-8968-7. Epub 2018 Jul 13.
4
From gene to therapy in spinal and bulbar muscular atrophy: Are we there yet?从脊髓性肌萎缩症的基因到治疗:我们做到了吗?
Mol Cell Endocrinol. 2018 Apr 15;465:113-121. doi: 10.1016/j.mce.2017.07.005. Epub 2017 Jul 5.
5
Nucleic Acid-Based Therapeutic Approach for Spinal and Bulbar Muscular Atrophy and Related Neurological Disorders.基于核酸的脊髓性肌萎缩症和相关神经疾病治疗方法。
Genes (Basel). 2022 Jan 5;13(1):109. doi: 10.3390/genes13010109.
6
Pathogenesis and molecular targeted therapy of spinal and bulbar muscular atrophy (SBMA).脊髓延髓肌萎缩症(SBMA)的发病机制和分子靶向治疗。
Cell Tissue Res. 2012 Jul;349(1):313-20. doi: 10.1007/s00441-012-1377-9. Epub 2012 Apr 4.
7
New routes to therapy for spinal and bulbar muscular atrophy.治疗脊髓性肌萎缩症和延髓性肌萎缩症的新途径。
J Mol Neurosci. 2013 Jul;50(3):514-23. doi: 10.1007/s12031-013-9978-7. Epub 2013 Feb 19.
8
Introduction to the Special Issue on Spinal and Bulbar Muscular Atrophy.脊髓延髓性肌萎缩专题介绍
J Mol Neurosci. 2016 Mar;58(3):313-6. doi: 10.1007/s12031-016-0720-0. Epub 2016 Feb 13.
9
Incidence and prevalence of Spinal and bulbar muscular atrophy in South Korea: a nationwide population-based study.韩国脊髓性和球部肌萎缩症的发病率和患病率:一项全国性基于人群的研究。
J Neurol. 2023 Oct;270(10):5017-5022. doi: 10.1007/s00415-023-11842-8. Epub 2023 Jun 30.
10
Metabolic alterations in spinal and bulbar muscular atrophy.脊髓性肌萎缩和延髓性肌萎缩的代谢改变。
Rev Neurol (Paris). 2020 Dec;176(10):780-787. doi: 10.1016/j.neurol.2020.03.020. Epub 2020 Jul 4.

引用本文的文献

1
Polyglutamine (PolyQ) Diseases: Navigating the Landscape of Neurodegeneration.多聚谷氨酰胺(PolyQ)疾病:探索神经退行性变的全景。
ACS Chem Neurosci. 2024 Aug 7;15(15):2665-2694. doi: 10.1021/acschemneuro.4c00184. Epub 2024 Jul 12.
2
Higher than expected incident cases of spinal bulbar muscular atrophy in western Canada.加拿大西部脊髓延髓肌肉萎缩症的发病病例高于预期。
Brain. 2024 May 3;147(5):e43-e44. doi: 10.1093/brain/awae052.

本文引用的文献

1
A versatile, high-efficiency platform for CRISPR-based gene activation.一种基于 CRISPR 的基因激活的多功能、高效平台。
Nat Commun. 2023 Feb 17;14(1):902. doi: 10.1038/s41467-023-36452-w.
2
An Antisense Oligonucleotide-Loaded Blood-Brain Barrier Penetrable Nanoparticle Mediating Recruitment of Endogenous Neural Stem Cells for the Treatment of Parkinson's Disease.一种负载反义寡核苷酸的血脑屏障可穿透纳米颗粒,介导内源性神经干细胞募集用于帕金森病的治疗。
ACS Nano. 2023 Mar 14;17(5):4414-4432. doi: 10.1021/acsnano.2c09752. Epub 2023 Jan 23.
3
Development of DG9 peptide-conjugated single- and multi-exon skipping therapies for the treatment of Duchenne muscular dystrophy.
DG9 肽缀合的单exon 和多exon 跳跃治疗药物的开发,用于治疗杜氏肌营养不良症。
Proc Natl Acad Sci U S A. 2022 Mar 1;119(9). doi: 10.1073/pnas.2112546119.
4
Gene therapy with AR isoform 2 rescues spinal and bulbar muscular atrophy phenotype by modulating AR transcriptional activity.基因治疗用 AR 异构体 2 通过调节 AR 转录活性来挽救脊髓性肌萎缩和延髓性肌萎缩的表型。
Sci Adv. 2021 Aug 20;7(34). doi: 10.1126/sciadv.abi6896. Print 2021 Aug.
5
Delivering Antisense Oligonucleotides across the Blood-Brain Barrier by Tumor Cell-Derived Small Apoptotic Bodies.通过肿瘤细胞衍生的小凋亡体递送至血脑屏障的反义寡核苷酸。
Adv Sci (Weinh). 2021 May 4;8(13):2004929. doi: 10.1002/advs.202004929. eCollection 2021 Jul.
6
Highly Elevated Prevalence of Spinobulbar Muscular Atrophy in Indigenous Communities in Canada Due to a Founder Effect.由于奠基者效应,加拿大原住民社区脊髓延髓肌肉萎缩症的患病率极高。
Neurol Genet. 2021 Jul 7;7(4):e607. doi: 10.1212/NXG.0000000000000607. eCollection 2021 Aug.
7
The Challenges and Strategies of Antisense Oligonucleotide Drug Delivery.反义寡核苷酸药物递送的挑战与策略
Biomedicines. 2021 Apr 16;9(4):433. doi: 10.3390/biomedicines9040433.
8
Small Activating RNAs: Towards the Development of New Therapeutic Agents and Clinical Treatments.小激活 RNA:迈向新型治疗药物和临床治疗的发展。
Cells. 2021 Mar 8;10(3):591. doi: 10.3390/cells10030591.
9
Antisense technology: an overview and prospectus.反义技术:概述与展望。
Nat Rev Drug Discov. 2021 Jun;20(6):427-453. doi: 10.1038/s41573-021-00162-z. Epub 2021 Mar 24.
10
Development and Validation of CRISPR Activator Systems for Overexpression of CB1 Receptors in Neurons.用于在神经元中过表达CB1受体的CRISPR激活系统的开发与验证
Front Mol Neurosci. 2020 Sep 8;13:168. doi: 10.3389/fnmol.2020.00168. eCollection 2020.