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溶酶体贮积症的动物模型:猫黏多糖贮积症VI型的一例新病例。

Animal models for lysosomal storage diseases: a new case of feline mucopolysaccharidosis VI.

作者信息

Di Natale P, Annella T, Daniele A, Spagnuolo G, Cerundolo R, de Caprariis D, Gravino A E

机构信息

Dipartimento di Biochimica e Biotechnologie Mediche, II Facoltà di Medicina e Chirurgia, Università di Napoli, Italy.

出版信息

J Inherit Metab Dis. 1992;15(1):17-24. doi: 10.1007/BF01800339.

Abstract

Two long-haired Siamese cats are reported with clinical manifestations of human mucopolysaccharidosis VI (Maroteaux-Lamy disease): facial dysmorphia, dysostosis multiplex, paralysis. Urine of the two affected animals contained a high concentration of glycosaminoglycans, as detected by the dimethylmethylene blue test. Qualitative analysis, performed by thin-layer chromatography of the cetylpyridinium chloride-precipitable material, showed dermatan sulphate. Excessive incorporation of [35S]sulphate in the intracellular mucopolysaccharide of cultured fibroblasts and deficiency of arylsulphatase B in such cells indicate that these cats are affected by Maroteaux-Lamy disease. They should thus be considered the first European case of feline mucopolysaccharidosis VI.

摘要

据报道,两只长毛暹罗猫出现了人类黏多糖贮积症VI型(马罗-拉米病)的临床表现:面部畸形、多发性骨发育异常、瘫痪。通过二甲基亚甲基蓝试验检测,两只患病动物的尿液中含有高浓度的糖胺聚糖。对十六烷基吡啶氯化物可沉淀物质进行薄层色谱法的定性分析,结果显示为硫酸皮肤素。培养的成纤维细胞的细胞内黏多糖中[35S]硫酸盐的过度掺入以及这些细胞中芳基硫酸酯酶B的缺乏表明,这些猫患有马罗-拉米病。因此,它们应被视为欧洲首例猫黏多糖贮积症VI型病例。

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