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几种有或没有进行骨髓移植的贮积病的神经心理学转归

Neuropsychological outcomes of several storage diseases with and without bone marrow transplantation.

作者信息

Shapiro E G, Lockman L A, Balthazor M, Krivit W

机构信息

Department of Neurology, University of Minnesota, Minneapolis 55455, USA.

出版信息

J Inherit Metab Dis. 1995;18(4):413-29. doi: 10.1007/BF00710053.

Abstract

Neuropsychological assessment is essential in providing documentation of the untreated natural history of storage diseases associated with dementia and quantifying the effectiveness of treatment on central nervous system function. Baseline characterization and outcome of bone marrow transplantation (BMT) for three leukodystrophies and three mucopolysaccharidoses are presented. Results suggests that BMT for Hurler syndrome, adrenoleukodystrophy, and globoid cell leukodystrophy can be effective in preventing dementia if done early enough in the disease. Sanfilippo and Hunter syndromes do not benefit and BMT is not recommended. For metachromatic leukodystrophy, BMT is not recommended for symptomatic early-onset forms of the disease. Further longitudinal follow-up is needed to determine whether the benefits outweigh the risks of BMT for late-onset and preclinical metachromatic leukodystrophy.

摘要

神经心理学评估对于记录与痴呆相关的贮积病未经治疗的自然病程以及量化治疗对中枢神经系统功能的有效性至关重要。本文介绍了三种脑白质营养不良和三种黏多糖贮积症的骨髓移植(BMT)的基线特征和结果。结果表明,如果在疾病早期进行,针对Hurler综合征、肾上腺脑白质营养不良和球状细胞脑白质营养不良的BMT在预防痴呆方面可能有效。Sanfilippo综合征和Hunter综合征无益处,不建议进行BMT。对于异染性脑白质营养不良,不建议对有症状的早发型疾病形式进行BMT。需要进一步的纵向随访来确定BMT对于晚发型和临床前异染性脑白质营养不良的益处是否大于风险。

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