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雷特综合征:提示轴索性神经病和线粒体异常的发现。

Rett syndrome: findings suggesting axonopathy and mitochondrial abnormalities.

作者信息

Wakai S, Kameda K, Ishikawa Y, Miyamoto S, Nagaoka M, Okabe M, Minami R, Tachi N

机构信息

Department of Pediatrics, National Sanitorium Yakumo Hospital, Hokkaido, Japan.

出版信息

Pediatr Neurol. 1990 Sep-Oct;6(5):339-43. doi: 10.1016/0887-8994(90)90028-y.

Abstract

We report the histopathologic findings of 3 sural nerve biopsies and 1 muscle biopsy from 3 patients with Rett syndrome. The 3 sural nerve biopsies demonstrated a few ultrastructural abnormalities, including the presence of many Pi-granules and mitochondrial changes in the cytoplasm of Schwann cells, occasional bands of Büngner and onion-bulb formations, and mitochondrial alterations in myelinated axons. Morphometric analysis disclosed reduction in the number of large myelinated fibers with normal densities in comparison to those of an age-matched normal control. Light microscopic examination of the biopsied muscle from a 6-year-old patient with Rett syndrome revealed the existence of many small, dark, angulated fibers with NADH-TR staining. Ultrastructural investigation of the muscle confirmed the presence of the dumbbell-shaped mitochondria. Peripheral nerve involvement and the possibility of mitochondrial abnormalities in Rett syndrome were suggested by the results.

摘要

我们报告了3例雷特综合征患者的3次腓肠神经活检和1次肌肉活检的组织病理学结果。3次腓肠神经活检显示出一些超微结构异常,包括施万细胞胞质中存在许多P颗粒和线粒体变化、偶尔出现的Büngner带和洋葱球形成,以及有髓轴突中的线粒体改变。形态计量分析显示,与年龄匹配的正常对照相比,大的有髓纤维数量减少但密度正常。对一名6岁雷特综合征患者的活检肌肉进行光镜检查发现,经NADH-TR染色后存在许多小的、深色的、有角的纤维。对肌肉的超微结构研究证实了哑铃形线粒体的存在。这些结果提示雷特综合征存在周围神经受累以及线粒体异常的可能性。

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