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超越运动神经元:扩张肯尼迪病的临床谱。

Beyond motor neurons: expanding the clinical spectrum in Kennedy's disease.

机构信息

Department of Physiology, Anatomy and Genetics, University of Oxford, Oxford, UK.

Department of Neurosciences, Neuromuscular Center, University of Padova, Padova, Italy.

出版信息

J Neurol Neurosurg Psychiatry. 2018 Aug;89(8):808-812. doi: 10.1136/jnnp-2017-316961. Epub 2018 Jan 20.

Abstract

Kennedy's disease, or spinal and bulbar muscular atrophy (SBMA), is an X-linked neuromuscular condition clinically characterised by weakness, atrophy and fasciculations of the limb and bulbar muscles, as a result of lower motor neuron degeneration. The disease is caused by an abnormally expanded triplet repeat expansions in the ubiquitously expressed androgen receptor gene, through mechanisms which are not entirely elucidated. Over the years studies from both humans and animal models have highlighted the involvement of cell populations other than motor neurons in SBMA, widening the disease phenotype. The most compelling aspect of these findings is their potential for therapeutic impact: muscle, for example, which is primarily affected in the disease, has been recently shown to represent a valid alternative target for therapy to motor neurons. In this review, we discuss the emerging study of the extra-motor neuron involvement in SBMA, which, besides increasingly pointing towards a multidisciplinary approach for affected patients, deepens our understanding of the pathogenic mechanisms and holds potential for providing new therapeutic targets for this disease.

摘要

肯尼迪病,或脊髓延髓肌萎缩症(SBMA),是一种 X 连锁的神经肌肉疾病,临床上表现为四肢和球部肌肉无力、萎缩和肌束颤动,是由于下运动神经元退化所致。该病是由雄激素受体基因中普遍表达的三核苷酸重复扩展异常引起的,但具体机制尚未完全阐明。多年来,来自人类和动物模型的研究都强调了除运动神经元以外的细胞群体在 SBMA 中的参与,从而拓宽了疾病表型。这些发现最引人注目的方面是它们具有治疗影响的潜力:例如,受疾病主要影响的肌肉最近被证明是运动神经元以外的另一种有效治疗靶点。在这篇综述中,我们讨论了 SBMA 中运动神经元以外的细胞参与的新兴研究,这些研究除了越来越指向针对受影响患者的多学科方法外,还加深了我们对发病机制的理解,并为这种疾病提供了新的治疗靶点。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8801/6204939/003438b9e8e0/jnnp-2017-316961f01.jpg

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