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弗里德赖希共济失调:临床特征和新进展。

Friedreich ataxia: clinical features and new developments.

机构信息

Departments of Pediatrics & Neurology, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA 19104, USA.

出版信息

Neurodegener Dis Manag. 2022 Oct;12(5):267-283. doi: 10.2217/nmt-2022-0011. Epub 2022 Jun 29.

Abstract

Friedreich's ataxia (FRDA), a neurodegenerative disease characterized by ataxia and other neurological features, affects 1 in 50,000-100,000 individuals in the USA. However, FRDA also includes cardiac, orthopedic and endocrine dysfunction, giving rise to many secondary disease characteristics. The multifaceted approach for clinical care has necessitated the development of disease-specific clinical care guidelines. New developments in FRDA include the advancement of clinical drug trials targeting the NRF2 pathway and frataxin restoration. Additionally, a novel understanding of gene silencing in FRDA, reflecting a variegated silencing pattern, will have applications to current and future therapeutic interventions. Finally, new perspectives on the neuroanatomy of FRDA and its developmental features will refine the time course and anatomical targeting of novel approaches.

摘要

弗里德赖希共济失调(FRDA)是一种神经退行性疾病,以共济失调和其他神经特征为特征,在美国每 50,000 至 100,000 人中就有 1 人受到影响。然而,FRDA 还包括心脏、骨科和内分泌功能障碍,导致许多继发性疾病特征。为了进行多方面的临床护理,有必要制定针对特定疾病的临床护理指南。FRDA 的新进展包括针对 NRF2 途径和 frataxin 恢复的临床药物试验的进展。此外,对 FRDA 中基因沉默的新认识,反映出斑驳的沉默模式,将应用于当前和未来的治疗干预措施。最后,对 FRDA 的神经解剖结构及其发育特征的新观点将完善新方法的时间进程和解剖靶向性。

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