Suppr超能文献

成人发病型克拉伯病尸检病例报告:与婴儿发病型病例的比较。

An autopsy case report of adult-onset Krabbe disease: Comparison with an infantile-onset case.

机构信息

Department of Cancer Pathology, Faculty of Medicine, Hokkaido University, Sapporo, Japan.

Department of Neurology, Nakamura Memorial Hospital, Sapporo, Japan.

出版信息

Pathol Int. 2022 Nov;72(11):558-565. doi: 10.1111/pin.13275. Epub 2022 Oct 6.

Abstract

Krabbe disease is a lysosomal storage disease caused by a deficiency of the galactocerebrosidase (GALC) enzyme, which leads to demyelination of the central and peripheral nervous systems. Almost all patients with Krabbe disease are infants, and this is the first report of adult-onset cases that describe pathological findings. Here, we present two autopsy cases: a 73-year-old female and a 2-year-old male. The adult-onset case developed symptoms in her late thirties and was diagnosed by the identification of GALC D528N and L634S mutations and by T2-weighted magnetic resonance imaging; she had increased signal in the white matter along the pyramidal tract to the bilateral precentral gyrus, as well as from the triangular part to the posterior horn of the lateral ventricle. Microscopically, Klüver-Barrera staining was pale in the white matter of the precentral gyrus and occipito-thalamic radiation, and a few globoid cells were observed. The GALC mutations that were identified in the present adult-onset case do not completely inactivate GALC enzyme activity, resulting in focal demyelination of the brain.

摘要

克拉伯病是一种溶酶体贮积症,由半乳糖脑苷脂酶(GALC)缺乏引起,导致中枢和周围神经系统脱髓鞘。几乎所有克拉伯病患者都是婴儿,这是首例描述成人发病病例的病理发现报告。本文报道了 2 例尸检病例:1 例为 73 岁女性,1 例为 2 岁男性。成年发病患者在三十多岁后期出现症状,并通过 GALC D528N 和 L634S 突变的鉴定以及 T2 加权磁共振成像诊断;她在锥体束沿途的白质、双侧中央前回,以及从三角区到侧脑室后角处,信号增强。显微镜下,中央前回和枕叶-丘脑辐射的白质 Klüver-Barrera 染色苍白,观察到少数球样细胞。本成年发病病例中鉴定出的 GALC 突变并未完全使 GALC 酶活性失活,导致大脑局灶性脱髓鞘。

相似文献

2
A new compound heterozygous mutation in adult-onset Krabbe disease.成人型克拉伯病中的一种新的复合杂合突变。
Int J Neurosci. 2020 Dec;130(12):1267-1271. doi: 10.1080/00207454.2020.1731504. Epub 2020 Feb 25.

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验