Kissel J T, Beam W, Bresolin N, Gibbons G, DiMauro S, Mendell J R
Neurology. 1985 Jun;35(6):828-33. doi: 10.1212/wnl.35.6.828.
A third case of phosphoglycerate mutase (PGAM) deficiency, a metabolic myopathy involving terminal glycolysis, was identified in a 24-year-old black man with episodic, exercise-induced myoglobinuria since age 13. To better understand the physiologic consequences of PGAM deficiency, incremental exercise testing was performed. Results were compared with those of two patients having myophosphorylase deficiency and five normals. In contrast to the patients with phosphorylase deficiency, the PGAM-deficient patient achieved near-normal levels of maximal exercise and produced a normal peak lactate after exercise. The mechanisms underlying the asymptomatic performance of such strenuous exercise in this case are uncertain, but the data suggest that unidentified factors are operative in precipitating attacks of myoglobinuria in patients with some metabolic myopathies. Despite similar clinical histories, patients with different glycolytic enzyme deficiencies can have striking differences in exercise tolerance.
第三例磷酸甘油酸变位酶(PGAM)缺乏症患者被确诊,这是一种涉及糖酵解终末阶段的代谢性肌病。该患者为一名24岁黑人男性,自13岁起就患有发作性运动诱发的肌红蛋白尿。为了更好地理解PGAM缺乏症的生理后果,进行了递增运动试验。结果与两名肌磷酸化酶缺乏症患者及五名正常人的结果进行了比较。与磷酸化酶缺乏症患者不同,PGAM缺乏症患者的最大运动量接近正常水平,运动后产生的乳酸峰值也正常。在这种情况下,如此剧烈运动却无症状表现的潜在机制尚不清楚,但数据表明,在某些代谢性肌病患者中,存在一些不明因素会促使肌红蛋白尿发作。尽管临床病史相似,但不同糖酵解酶缺乏症患者的运动耐量可能存在显著差异。