Sansone V, Rotondo G, Bottiroli G, Tremblay J P, Meola G
Division of Neurology II, University of Milan, San Donato Hospital, Italy.
Eur J Histochem. 1993;37(3):241-8.
In previous experiments, we obtained an in vitro restoration of a cytosolic enzyme glucose-6-phosphate dehydrogenase (G6PD) in hybrid myotubes formed between G6PD-deficient and human or murine myoblasts. In the present series of experiments, the degree of restoration was observed and the persistence of the restored activity was evaluated in the hybrids formed. Quantitative measurements of enzymatic activity in single human-human or human-mouse myotubes, identified by using either fluorescent latex microspheres or Hoechst stain 33258, were made by using a computer-controlled Leitz photometer. Histospectrophotometry and statistical analysis showed a 50% restoration of enzymatic activity in human-human hybrids compared to deficient and normal myotubes. The restored activity was uniformly distributed throughout the cytoplasm and persisted in long-term cultures. No nuclear domain was observed for G6PD. Knowledge of the degree of restoration, of the extent of distribution of the products of "competent" nuclei and the demonstration that the correction is not a transitory event in vitro, supports the potential usefulness of myoblast transfer therapy for this type of myopathy.
在先前的实验中,我们在葡萄糖-6-磷酸脱氢酶(G6PD)缺陷型与人类或鼠类成肌细胞形成的杂种肌管中实现了一种胞质酶的体外恢复。在本系列实验中,观察了恢复程度,并评估了所形成杂种中恢复活性的持久性。通过使用荧光乳胶微球或Hoechst 33258染色鉴定的单个人类-人类或人类-小鼠肌管中的酶活性定量测量,是使用计算机控制的Leitz光度计进行的。组织分光光度法和统计分析表明,与缺陷型和正常肌管相比,人类-人类杂种中的酶活性恢复了50%。恢复的活性均匀分布于整个细胞质中,并在长期培养中持续存在。未观察到G6PD的核区域。了解恢复程度、“有功能”细胞核产物的分布范围以及证明这种校正在体外不是一个短暂事件,支持了成肌细胞移植疗法对这类肌病的潜在有用性。