Tahmoush A J, Askanas V, Nelson P G, Engel W K
Arch Neurol. 1984 Nov;41(11):1190-2. doi: 10.1001/archneur.1984.04050220088024.
Electrophysiological studies were performed on aneurally cultured muscle cells from one patient with adult-onset acid maltase deficiency (AAMD) and from controls. The cells from the patient with AAMD had a higher mean resting membrane potential, a lower input resistance, and a higher incidence of action potentials at resting membrane potential than the control cells. Therefore, sarcolemma maturation was not adversely affected. The AAMD cells had membrane thresholds and action potential amplitudes similar to those of the control cells, and rarely produced repetitive action potentials. Therefore, the membrane instability noted in adult muscle fibers from the patient with AAMD was not present in cultured cells. This study does not support the suggestion that the biochemical and morphological abnormalities present in muscle fibers of patients with AAMD are sufficient to cause the electrical abnormalities.
对一名成年起病的酸性麦芽糖酶缺乏症(AAMD)患者和对照组的去神经培养肌细胞进行了电生理研究。与对照细胞相比,AAMD患者的细胞具有更高的平均静息膜电位、更低的输入电阻以及在静息膜电位时更高的动作电位发生率。因此,肌膜成熟未受到不利影响。AAMD细胞的膜阈值和动作电位幅度与对照细胞相似,且很少产生重复动作电位。因此,AAMD患者成年肌纤维中出现的膜不稳定在培养细胞中不存在。本研究不支持以下观点,即AAMD患者肌纤维中存在的生化和形态学异常足以导致电异常。