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脊髓性肌萎缩症:不仅仅是运动神经元疾病?

Spinal Muscular Atrophy: More than a Disease of Motor Neurons?

作者信息

Nash L A, Burns J K, Chardon J Warman, Kothary R, Parks R J

机构信息

Regenerative Medicine Program, Ottawa Hospital Research Institute, General Campus Général, 501 Smyth Road, Room C4415, Ottawa, ON K1H 8L6, Canada.

出版信息

Curr Mol Med. 2016;16(9):779-792. doi: 10.2174/1566524016666161128113338.

Abstract

Spinal muscular atrophy (SMA) is the most common genetically inherited neurodegenerative disease resulting in infant mortality. SMA is caused by genetic deletion or mutation in the survival of motor neuron 1 (SMN1) gene, which results in reduced levels of the survival of motor neuron (SMN) protein. SMN protein deficiency preferentially affects α- motor neurons, leading to their degeneration and subsequent atrophy of limb and trunk muscles, progressing to death in severe forms of the disease. More recent studies have shown that SMN protein depletion is detrimental to the functioning of other tissues including skeletal muscle, heart, autonomic and enteric nervous systems, metabolic/endocrine (e.g. pancreas), lymphatic, bone and reproductive system. In this review, we summarize studies discussing SMN protein's function in various cell and tissue types and their involvement in the context of SMA disease etiology. Taken together, these studies indicate that SMA is a multi-organ disease, which suggests that truly effective disease intervention may require body-wide correction of SMN protein levels.

摘要

脊髓性肌萎缩症(SMA)是导致婴儿死亡的最常见的遗传性神经退行性疾病。SMA由运动神经元存活1(SMN1)基因的遗传缺失或突变引起,这导致运动神经元存活(SMN)蛋白水平降低。SMN蛋白缺乏优先影响α运动神经元,导致其退化以及随后肢体和躯干肌肉萎缩,在疾病的严重形式中会发展至死亡。最近的研究表明,SMN蛋白耗竭对包括骨骼肌、心脏、自主神经系统和肠神经系统、代谢/内分泌系统(如胰腺)、淋巴系统、骨骼和生殖系统在内的其他组织的功能有害。在本综述中,我们总结了讨论SMN蛋白在各种细胞和组织类型中的功能及其在SMA疾病病因学背景下所涉及情况的研究。综上所述,这些研究表明SMA是一种多器官疾病,这表明真正有效的疾病干预可能需要全身性纠正SMN蛋白水平。

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