Godin D V, Herring F G, MacLeod P J
J Med. 1981;12(1):35-49.
Structural and functional characteristics of erythrocytes and isolated erythrocyte membranes from known malignant hyperthermia (MH) carriers have been examined in the hope of deriving some information concerning the underlying molecular basis of this genetic abnormality, which may represent a state of generalized membrane involvement. The increase in erythrocyte osmotic fragility which has previously been noted in porcine MH was found not to apply to the human disorder and there was evidence that in some individuals at risk osmotic fragility was in fact reduced. Although no alteration in erythrocyte membrane phospholipid profiles was detected, membrane cholesterol levels were reduced in all three definite carriers examined as well as in approximately half of the possible MH carriers investigated. No evidence for associated changes in membrane protein sulfhydryl group latency or in temperature-dependent perturbations of membrane fluidity using a stearic acid spin probe could be detected. Finally, since alterations at the level of skeletal muscle membrane -Ca++ interaction have been implicated in the pathogenesis of MH, we have examined in detail the influence of temperature on the Ca++-stimulated components of the Mg++-dependent ATPase of erythrocyte membranes from known MH carriers but no evidence of any abnormality could be found. Since MH carriers detection based solely on measurements of plasma creatine phosphokinase elevations may yield equivocal results, a decrease in erythrocyte membrane cholesterol content may provide a convenient means of identifying such individuals at risk.
已对已知恶性高热(MH)携带者的红细胞及分离出的红细胞膜的结构和功能特征进行了研究,以期获得一些有关这种基因异常潜在分子基础的信息,这种异常可能代表一种广泛的膜受累状态。先前在猪MH中发现的红细胞渗透脆性增加在人类疾病中并不适用,且有证据表明,在一些有风险的个体中,渗透脆性实际上降低了。虽然未检测到红细胞膜磷脂谱的改变,但在所检测的所有三名确诊携带者以及约一半被调查的可能MH携带者中,膜胆固醇水平均降低。使用硬脂酸自旋探针未检测到膜蛋白巯基潜伏性或膜流动性温度依赖性扰动的相关变化。最后,由于骨骼肌膜 -Ca++ 相互作用水平的改变与MH的发病机制有关,我们详细研究了温度对已知MH携带者红细胞膜Mg++ 依赖性ATP酶的Ca++ 刺激成分的影响,但未发现任何异常迹象。由于仅基于血浆肌酸磷酸激酶升高的测量来检测MH携带者可能会产生模棱两可的结果,红细胞膜胆固醇含量的降低可能为识别此类有风险个体提供一种便捷方法。