Kost G J, Verity M A
Muscle Nerve. 1980 May-Jun;3(3):195-201. doi: 10.1002/mus.880030302.
McArdle disease classically presents in childhood or adolescence. Rarely does it become symptomatic for the first time in late adulthood, with the onset of progressive muscle wasting and weakness. Our patient is unusual in that despite a life of physical vigor, she developed immobilizing cramps, stiffness, and muscle swelling abruptly at age 60. She had no previous symptoms of muscle disease. The diagnosis was indicated by the ischemic forearm test, which produced muscle contracture and no rise in venous lactate levels, and confirmed by histochemical, electrophoretic, and biochemical studies that showed complete absence of myophosphorylase. This case defines a new variant of the late-onset type and raises important questions about compensatory mechanisms, inheritance patterns, and etiological factors in myophosphorylase deficiency.
麦克尔迪氏病通常在儿童期或青少年期发病。在成年晚期首次出现症状,表现为进行性肌肉萎缩和无力的情况极为罕见。我们的患者不同寻常之处在于,尽管她一生体力充沛,但在60岁时突然出现了导致活动受限的痉挛、僵硬和肌肉肿胀。她此前没有肌肉疾病的症状。缺血性前臂试验显示肌肉挛缩且静脉乳酸水平未升高,提示了诊断,组织化学、电泳和生化研究证实完全缺乏肌磷酸化酶,从而确诊。该病例定义了迟发型的一种新变体,并引发了关于肌磷酸化酶缺乏的代偿机制、遗传模式和病因学因素的重要问题。