Yoshida H, Kuriyama M
Department of Pathology, Faculty of Medicine, Kagoshima University, Japan.
Lab Anim Sci. 1990 Sep;40(5):486-9.
We describe a new animal model of a genetic lipid storage disease analogous to human Wolman's disease. Affected Donryu rats, who inherited the disease in an autosomal recessive mode, manifested marked hepatosplenomegaly, lymph node enlargement, and thickened, dilated intestine. Morphologically, many characteristic foam cells were observed in livers and spleens. No adrenal calcification could be found in affected rats. Biochemical studies on spleen and liver tissues showed massive accumulation of esterified cholesterol and triglycerides, and deficiency of acid lipase for [14C]-cholesteryl oleate. This animal model could contribute greatly to the clarification of the physiological and pathological roles of lysosomal acid lipase in the metabolism of lipoproteins and cholesterol, and of the pathogenesis of atherosclerosis.
我们描述了一种类似于人类沃尔曼病的遗传性脂质贮积病的新动物模型。患病的唐育大鼠以常染色体隐性模式遗传该病,表现出明显的肝脾肿大、淋巴结肿大以及肠壁增厚和扩张。形态学上,在肝脏和脾脏中观察到许多特征性的泡沫细胞。患病大鼠未发现肾上腺钙化。对脾脏和肝脏组织的生化研究表明,酯化胆固醇和甘油三酯大量蓄积,且缺乏针对[14C]-胆固醇油酸酯的酸性脂肪酶。这种动物模型对于阐明溶酶体酸性脂肪酶在脂蛋白和胆固醇代谢中的生理和病理作用以及动脉粥样硬化的发病机制可能有很大帮助。