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肉碱水平正常的脂质贮积性肌病

Lipid storage myopathy with normal carnitine levels.

作者信息

Jerusalem F, Spiess H, Baumgartner G

出版信息

J Neurol Sci. 1975 Mar;24(3):273-82. doi: 10.1016/0022-510x(75)90247-6.

Abstract

A 28-year-old female, who showed a floppy baby syndrome during early infancy, had a non-progressive proximal muscle weakness with easy fatiguability since childhood. Two muscle specimens biopsied at the age of 28 years revealed myriads of 1-3-mum wide abnormal spaces containing neutral fat in type I and type II fibers. Both biopsies demonstrated a type I fiber preponderance. Electron microscopy demonstrated lipid excess and normal mitochondria by simple inspection. The mitochondrial area and sarcotubular membrane profile concentration in morphometry of longitudinal sections were also normal. Cross-sections, however, revealed a slight decrease of the individual mitochondrial size and of the sarcotubular membrane profile concentration . Serum and muscle carnitine levels and the muscle carnitine palmityltransferase level were all within normal range. Besides carnitine deficiency other biochemical defects can occur in lipid storage myopathy, which represents a syndrome rather than a unique disease entity.

摘要

一名28岁女性,在婴儿早期表现出松软婴儿综合征,自童年起就有非进行性近端肌无力且易疲劳。28岁时取的两块肌肉活检标本显示,I型和II型纤维中有无数1 - 3微米宽的异常间隙,内含中性脂肪。两次活检均显示I型纤维占优势。电子显微镜检查通过简单观察显示脂质过多且线粒体正常。纵切面形态计量学中的线粒体面积和肌管膜轮廓浓度也正常。然而,横切面显示单个线粒体大小和肌管膜轮廓浓度略有下降。血清和肌肉肉碱水平以及肌肉肉碱棕榈酰转移酶水平均在正常范围内。除了肉碱缺乏外,脂质贮积性肌病还可能出现其他生化缺陷,脂质贮积性肌病代表一种综合征而非单一的疾病实体。

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