Grebner E E, Mansfield D A, Raghavan S S, Kolodny E H, d'Azzo A, Neufeld E F, Jackson L G
Am J Hum Genet. 1986 Apr;38(4):505-14.
Two abnormalities of beta-hexosaminidase A (HEX A) activity are described. One, found in two unrelated Jewish children, was characterized by the complete absence of HEX A activity in serum, but low levels of activity in leukocytes and fibroblasts using artificial substrate. The other, found in a non-Jewish man, was characterized by uniformly low levels of HEX A activity in leukocytes, fibroblasts, and serum against artificial substrate. In all cases, the pH optimum of HEX A was normal, there was no increased lability at 37 degrees C, and no inhibitor was detected to account for the deficiency of activity. Cultured fibroblasts of these individuals were capable of synthesizing and processing alpha- and beta-subunits of HEX A and capable of cleaving GM2 ganglioside. The patients, ranging in age from 6 to 30 years, are clinically normal. They are probably genetic compounds carrying the classical Tay-Sachs gene and a differently mutated allele that imparts the anomalous phenotypic features observed.
描述了β-己糖胺酶A(HEX A)活性的两种异常情况。一种情况见于两名无血缘关系的犹太儿童,其特征是血清中完全缺乏HEX A活性,但使用人工底物时白细胞和成纤维细胞中的活性水平较低。另一种情况见于一名非犹太男性,其特征是白细胞、成纤维细胞和血清中针对人工底物的HEX A活性均持续处于低水平。在所有病例中,HEX A的最适pH值正常,在37℃时稳定性未增加,且未检测到抑制剂可解释活性缺乏的原因。这些个体的培养成纤维细胞能够合成和加工HEX A的α亚基和β亚基,并且能够裂解GM2神经节苷脂。患者年龄在6至30岁之间,临床检查正常。他们可能是携带经典泰-萨克斯基因和另一个不同突变等位基因的遗传复合杂合子,该等位基因赋予了所观察到的异常表型特征。