Argov Z, Navon R
Ann Neurol. 1984 Jul;16(1):14-20. doi: 10.1002/ana.410160105.
Six patients from three families developed adult GM2 gangliosidosis resulting from severe beta-hexosaminidase A deficiency. The clinical picture varied between and within families and included spinocerebellar, various motor neuron, and cerebellar connection syndromes. Psychosis appeared in each family. Involvement of three generations was recorded in one family. The phenotype of adult GM2 gangliosidosis is variable and cannot form a basis for genetic classification. Detailed hexosaminidase determinations in eight patients from five families revealed all patients to have minor quantities of hexosaminidase A (about 15% of normal), with marked increase in hexosaminidase I isozyme.
来自三个家族的6名患者因严重的β-己糖胺酶A缺乏而患上成人GM2神经节苷脂贮积症。不同家族之间以及家族内部的临床表现各不相同,包括脊髓小脑、各种运动神经元和小脑连接综合征。每个家族中都出现了精神病症状。在一个家族中记录到了三代人受累的情况。成人GM2神经节苷脂贮积症的表型具有多样性,不能作为遗传分类的依据。对来自五个家族的8名患者进行的详细己糖胺酶测定显示,所有患者的己糖胺酶A含量均较少(约为正常水平的15%),而己糖胺酶I同工酶显著增加。