Pentchev P G, Gal A E, Boothe A D, Fouks J, Omodeo-Sale F, Brady R O
Birth Defects Orig Artic Ser. 1980;16(1):225-30.
A strain of BALB/c mice with an autosomal recessive neurologic disorder has been reported previously [1, 2]. The tissues of affected animals have been further examined and the activities of varius lysosomal hydrolases and levels of sphingolipids were compared to those in control mice. There was a substantial diminution of sphingomyelinase and glucocerebrosidase activities in liver, spleen, lung, thymus, and kidney of affected mice. There was a corresponding accumulation of sphingomyelin and glucocerebroside in these tissues. The activity of several other lysosomal hydrolases was elevated. Heterozygotes did not show any of the enzymatic alterations. The brain of affected animals showed substantial accumulation of the gangliosides GM3 and GM2.
先前已报道过一种患有常染色体隐性神经疾病的BALB/c小鼠品系[1, 2]。已对患病动物的组织进行了进一步检查,并将各种溶酶体水解酶的活性和鞘脂水平与对照小鼠进行了比较。患病小鼠的肝脏、脾脏、肺、胸腺和肾脏中的鞘磷脂酶和葡萄糖脑苷脂酶活性大幅降低。这些组织中相应地积累了鞘磷脂和葡萄糖脑苷脂。其他几种溶酶体水解酶的活性升高。杂合子未表现出任何酶促改变。患病动物的大脑中神经节苷脂GM3和GM2大量积累。