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[肌聚糖病:最新进展与治疗前景]

[Sarcoglycanopathies: state of the art and therapeutic perspectives].

作者信息

Fernández-Eulate Gorka, Leturcq France, Laforêt Pascal, Richard Isabelle, Stojkovic Tanya

机构信息

Centre de Référence des maladies neuromusculaires Nord/Est/Île-de-France, APHP, Groupe Hospitalier Pitié-Salpêtrière, Sorbonne Université, Paris, France.

Laboratoire de biochimie génétique. APHP, Hôpital Cochin, Paris, France.

出版信息

Med Sci (Paris). 2020 Dec;36 Hors série n° 2:22-27. doi: 10.1051/medsci/2020243. Epub 2021 Jan 11.

Abstract

Sarcoglycanopathies are the third most common cause of autosomal recessive limb girdle muscular dystrophies (LGMD). They are the result of a deficiency in one of the sarcoglycans a, b, g, or d. The usual clinical presentation is that of a symmetrical involvement of the muscles of the pelvic and scapular girdles as well as of the trunk, associated with more or less severe cardio-respiratory impairment and a marked increase of serum CK levels. The first symptoms appear during the first decade, the loss of ambulation occurring often during the second decade. Lesions observed on the muscle biopsy are dystrophic. This is associated with a decrease or an absence of immunostaining of the sarcoglycan corresponding to the mutated gene and, to a lesser degree, of the other three sarcoglycans. Many mutations have been reported in the four incriminated genes and some of them are prevalent in certain populations. To date, there is no curative treatment, which does not prevent the development of many clinical trials, especially in gene therapy.

摘要

肌聚糖病是常染色体隐性遗传性肢带型肌营养不良症(LGMD)的第三大常见病因。它们是由于肌聚糖α、β、γ或δ中的一种缺乏所致。通常的临床表现是骨盆带和肩胛带以及躯干肌肉的对称性受累,伴有或多或少严重的心肺功能损害以及血清肌酸激酶(CK)水平显著升高。最初症状出现在第一个十年,常常在第二个十年出现行走能力丧失。肌肉活检观察到的病变为营养不良性。这与对应于突变基因的肌聚糖免疫染色减少或缺失相关,在较小程度上也与其他三种肌聚糖的免疫染色减少或缺失相关。在这四个致病基因中已报道了许多突变,其中一些在特定人群中较为普遍。迄今为止,尚无治愈性治疗方法,但这并未阻止许多临床试验的开展,尤其是基因治疗方面的试验。

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