Alroy J, Castagnaro M, Skutelsky E, Lomakina I
Department of Pathology, Tufts University School of Medicine, Boston, MA 02111.
Acta Neuropathol. 1994;87(6):594-7. doi: 10.1007/BF00293320.
In infantile lysosomal storage disease associated with osteopetrosis the nature of the enzyme deficiency as well as the type of material accumulated are both unknown. We used lectin histochemistry to characterize the storage material of previously reported cases. Using paraffin sections neurons stained positively with Luxol fast blue (LFB), periodic acid-Schiff (PAS), Concanavalia ensiformis agglutinin, Datura stramonium agglutinin, Griffonia simplicifolia-I, Lens culinaris agglutinin, Ricinus communis agglutinin-I, succinylated wheat germ agglutinin and wheat germ agglutinin, indicating an accumulation of fucosylated N-glycosidically linked oligosaccharides containing beta- and alpha-galactosyl residues and compounds containing N-acetyllactosamine. Reticuloendothelial cells in liver and in spleen did not stain with LFB, but did stain with PAS and the above lectins. These results indicate that there is storage of both carbohydrates and lipids in neurons, and stored carbohydrates with similar residues in reticuloendothelial cells in this disease, where the primary defect is still unknown.
在与骨质石化症相关的婴儿溶酶体贮积病中,酶缺乏的性质以及蓄积物质的类型均不明。我们采用凝集素组织化学法对先前报道病例的贮积物质进行特征分析。使用石蜡切片,神经元对卢戈氏坚牢蓝(LFB)、过碘酸希夫(PAS)、刀豆凝集素、曼陀罗凝集素、西非豆凝集素-I、菜豆凝集素、蓖麻凝集素-I、琥珀酰化麦胚凝集素和麦胚凝集素呈阳性染色,表明存在含有β-和α-半乳糖基残基的岩藻糖基化N-糖苷连接寡糖以及含有N-乙酰乳糖胺的化合物的蓄积。肝脏和脾脏中的网状内皮细胞对LFB不着色,但对PAS和上述凝集素着色。这些结果表明,在该疾病中,神经元中存在碳水化合物和脂质的蓄积,网状内皮细胞中存在具有相似残基的蓄积碳水化合物,而其主要缺陷仍然不明。