Bertagnolio B, Di Donato S, Peluchetti D, Rimoldi M, Storchi G, Cornelio F
Eur Neurol. 1978;17(4):193-204. doi: 10.1159/000114945.
3 adult women with distinct clinical pictures of progressive myopathy were studied. The morphological findings of biopsied skeletal muscle suggested the diagnosis of type II glycogenosis. Biochemical analysis confirmed a profound deficiency of alpha-1,4-glucosidase activity. Electrophoresis of muscle acid maltase showed the presence of one band in normal individuals. A very faint band with normal electrophoretic mobility was present in the patients' muscles. Muscle neutral maltase is composed of four bands in normal adult individuals: two of the four bands were clearly reduced in the muscles of the patients. The acid and neutral maltases were not significantly reduced in the patients' leukocytes. Acid maltase determination in urine made it possible to identify the homozygous, but not to completely segregate the heterozygous, from unaffected adult subjects.
对3名患有进行性肌病不同临床表现的成年女性进行了研究。活检骨骼肌的形态学结果提示诊断为II型糖原贮积症。生化分析证实α-1,4-葡萄糖苷酶活性严重缺乏。肌肉酸性麦芽糖酶的电泳显示正常个体中有一条带。患者肌肉中存在一条电泳迁移率正常但非常微弱的带。正常成年个体的肌肉中性麦芽糖酶由四条带组成:患者肌肉中四条带中的两条明显减少。患者白细胞中的酸性和中性麦芽糖酶没有明显减少。尿液中酸性麦芽糖酶的测定能够识别纯合子,但不能将杂合子与未受影响的成年受试者完全区分开来。