Meerbach W, Gräbner R
Zentralbl Allg Pathol. 1977;121(1-2):169-76.
A sporadic case of central core disease in a 5 1/2-year-old girl is reported. Clinically, a retarded motor development existed, furthermore, a muscle weakness and hypotonia of the extremities and trunk, contractures of the hip- and knee-joint,and luxation of both hip-joints. Biopsy specimens are taken from both Mm. gastrocnemii. Muscle fibres show, by morphologic examination, 95 per cent cores, which are characteristic for this myopathy. A further abnormality is seen inthe histochemical preparations for phosphorylase, succinate dehydrogenase, NAD diaphorase tetrazolium reductase, myofibrillar ATPase as well as AS-reaction with and without diastase digestion. With these techniques the muscle fibres show an uniform reaction pattern in which the activities of the oxidative andglycolytic enzymes correspond to the type I fibres of healthy persons. The cores show a lack of a activity of the oxidative and glycolytic enzymes as well as are ATPase- and PAS-negative. By reason of this histochemical behaviour it is suggested that the cores are predominantly unstructured. The cause of this disease might be complex disturbances in the neuro-muscular system manifested in the fetal period.
报告了一例5岁半女孩的散发性中央轴空病病例。临床上,存在运动发育迟缓,此外还有四肢和躯干肌肉无力、肌张力减退、髋关节和膝关节挛缩以及双侧髋关节脱位。从双侧腓肠肌取材进行活检。通过形态学检查,肌肉纤维显示95%为轴空,这是这种肌病的特征。在磷酸化酶、琥珀酸脱氢酶、NAD黄递酶四唑还原酶、肌原纤维ATP酶以及有无淀粉酶消化的AS反应的组织化学制剂中还发现了另一种异常。通过这些技术,肌肉纤维显示出一致的反应模式,其中氧化酶和糖酵解酶的活性与健康人的I型纤维相对应。轴空显示氧化酶和糖酵解酶缺乏活性,并且ATP酶和PAS呈阴性。基于这种组织化学表现,提示轴空主要是无结构的。这种疾病的病因可能是胎儿期神经肌肉系统的复杂紊乱。