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一名患有晚发性婴儿型异染性脑白质营养不良的5岁男童:病例报告。

A 5-year-old male child with late infantile metachromatic leukodystrophy: a case report.

作者信息

Mahmood Afreen, Chacham Swathi, Reddy Uppin Narayan, Rao Jillalla Narsing, Rao S Pratap

机构信息

Department of Paediatrics, Deccan College of Medical Sciences, Princess Esra Hospital, Hyderabad, Andhra Pradesh, India

Department of Paediatrics, Deccan College of Medical Sciences, Princess Esra Hospital, Hyderabad, Andhra Pradesh, India.

出版信息

J Child Neurol. 2015 Mar;30(4):483-5. doi: 10.1177/0883073814542948. Epub 2014 Aug 12.

Abstract

Metachromatic leukodystrophy is a rare disorder of myelin metabolism. This degenerative disorder results from the accumulation of cerebroside sulfatide within the myelin sheath of central and peripheral nervous system, due to deficiency of aryl sulfatase A enzyme. We report a 5-year-old male child, who presented with regression of milestones, recurrent seizures and spasticity from second year of life. Initially neurodegenerative disorder was considered and the case was investigated with neuroimaging and enzyme levels. Computed tomography (CT) of the brain showed hypodensities in the corpus callosum and bilateral periventricular and deep cerebral white matter suggestive of neurodegenerative disorder. Subsequently, magnetic resonance imaging (MRI) of the brain was done, which showed symmetrical hyperintensities in the periventricular white matter with classical sparing of subcortical "U" fibers. The β-galactosidase enzyme activity was normal; however, the activity of aryl sulfatase A enzyme was undetectable, confirming the diagnosis of late infantile variant of metachromatic leukodystrophy.

摘要

异染性脑白质营养不良是一种罕见的髓鞘代谢紊乱疾病。这种退行性疾病是由于芳基硫酸酯酶A酶缺乏,导致脑硫脂在中枢和周围神经系统的髓鞘内蓄积所致。我们报告一名5岁男童,自生命第二年起出现发育里程碑倒退、反复发作的癫痫和痉挛。最初考虑为神经退行性疾病,并通过神经影像学和酶水平进行了检查。脑部计算机断层扫描(CT)显示胼胝体以及双侧脑室周围和深部脑白质低密度,提示神经退行性疾病。随后进行了脑部磁共振成像(MRI),结果显示脑室周围白质对称高信号,皮质下“U”纤维典型性 spared。β-半乳糖苷酶活性正常;然而,芳基硫酸酯酶A酶活性检测不到,确诊为异染性脑白质营养不良晚婴型。

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