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一只患有桑德霍夫病犬的两年随访磁共振成像和波谱分析结果及脑脊液分析

Two-Year Follow-Up Magnetic Resonance Imaging and Spectroscopy Findings and Cerebrospinal Fluid Analysis of a Dog with Sandhoff's Disease.

作者信息

Ito D, Ishikawa C, Jeffery N D, Ono K, Tsuboi M, Uchida K, Yamato O, Kitagawa M

机构信息

School of Veterinary Medicine, Nihon University, Kanagawa, Japan.

Veterinary Medicine and Biomedical Sciences, Texas A&M University, TX.

出版信息

J Vet Intern Med. 2018 Mar;32(2):797-804. doi: 10.1111/jvim.15041. Epub 2018 Feb 25.

Abstract

A 13-month-old female Toy Poodle was presented for progressive ataxia and intention tremors of head movement. The diagnosis of Sandhoff's disease (GM2 gangliosidosis) was confirmed by deficient β-N-acetylhexosaminidase A and B activity in circulating leukocytes and identification of the homozygous mutation (HEXB: c.283delG). White matter in the cerebrum and cerebellum was hyperintense on T2-weighted and fluid-attenuated inversion recovery magnetic resonance images. Over the next 2 years, the white matter lesions expanded, and bilateral lesions appeared in the cerebellum and thalamus, associated with clinical deterioration. Magnetic resonance spectroscopy showed progressive decrease in brain N-acetylaspartate, and glycine-myo-inositol and lactate-alanine were increased in the terminal clinical stage. The concentrations of myelin basic protein and neuron specific enolase in cerebrospinal fluid were persistently increased. Imaging and spectroscopic appearance correlated with histopathological findings of severe myelin loss in cerebral and cerebellar white matter and destruction of the majority of cerebral and cerebellar neurons.

摘要

一只13个月大的雌性玩具贵宾犬因进行性共济失调和头部运动意向性震颤前来就诊。通过循环白细胞中β-N-乙酰己糖胺酶A和B活性缺乏以及纯合突变(HEXB:c.283delG)的鉴定,确诊为桑德霍夫病(GM2神经节苷脂沉积症)。在T2加权和液体衰减反转恢复磁共振图像上,大脑和小脑的白质呈高信号。在接下来的2年里,白质病变扩大,小脑和丘脑出现双侧病变,伴有临床恶化。磁共振波谱显示脑内N-乙酰天门冬氨酸逐渐减少,在临床终末期甘氨酸-肌醇和乳酸-丙氨酸增加。脑脊液中髓鞘碱性蛋白和神经元特异性烯醇化酶的浓度持续升高。影像学和波谱表现与大脑和小脑白质严重脱髓鞘以及大多数大脑和小脑神经元破坏的组织病理学结果相关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1fcc/5867010/a0f2ddc84908/JVIM-32-797-g001.jpg

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