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中央轴空病——一例报告

Central core disease--a case report.

作者信息

Myong N H, Suh Y L, Chi J G, Hwang Y S

机构信息

Department of Pathology, Seoul National University, Korea.

出版信息

J Korean Med Sci. 1993 Jun;8(3):235-40. doi: 10.3346/jkms.1993.8.3.235.

Abstract

Central core disease is a rare congenital myopathy characterized by the formation of "cores" that consist of abnormal arrangement of myofibrils inside the myofibers. We report a 5-year-old Korean girl who showed a fairly typical clinical course of non-progressive muscle weakness. Electrodiagnostic studies showed low-amplitude polyphasic electromyograph and normal nerve conduction velocity. Gastrocnemius muscle biopsy showed central cores in over 80% of the fibers on H&E section. Histochemistry revealed deficient or absent mitochondrial enzyme in the cores and type I predominance. Ultrastructurally both structured and non-structured cores were found separately or simultaneously in one fiber. This case is the first report in the Korean literature.

摘要

中央轴空病是一种罕见的先天性肌病,其特征是在肌纤维内形成由肌原纤维异常排列组成的“轴空”。我们报告一名5岁韩国女孩,其表现出相当典型的非进行性肌无力临床病程。电诊断研究显示肌电图为低波幅多相波,神经传导速度正常。腓肠肌活检显示苏木精-伊红(H&E)切片上超过80%的纤维有中央轴空。组织化学显示轴空内线粒体酶缺乏或缺失,且以I型为主。超微结构显示,在一根纤维中可单独或同时发现有结构和无结构的轴空。该病例是韩国文献中的首例报道。

相似文献

1
Central core disease--a case report.中央轴空病——一例报告
J Korean Med Sci. 1993 Jun;8(3):235-40. doi: 10.3346/jkms.1993.8.3.235.
2
Multicore myopathy--a case report.多核肌病——一例报告
J Korean Med Sci. 1993 Aug;8(4):312-7. doi: 10.3346/jkms.1993.8.4.312.
3
["Central core" myopathy: report of a case].["中央轴空" 性肌病:一例报告]
Arq Neuropsiquiatr. 1981 Jun;39(2):230-6. doi: 10.1590/s0004-282x1981000200012.
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Congenital fiber type disproportion: report of one case.先天性纤维类型比例失调:1例报告。
Zhonghua Min Guo Xiao Er Ke Yi Xue Hui Za Zhi. 1990 Nov-Dec;31(6):366-72.

引用本文的文献

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Core Disease in an Adult.成人核心疾病
Ann Indian Acad Neurol. 2022 Jul-Aug;25(4):729-731. doi: 10.4103/aian.aian_792_21. Epub 2022 Jul 14.

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