Izumi Rumiko, Suzuki Naoki, Kato Kazuhiro, Warita Hitoshi, Tateyama Maki, Nakashima Ichiro, Itoyama Yasuto
Department of Neurology, Tohoku University School of Medicine, Sendai.
Intern Med. 2010;49(15):1623-5. doi: 10.2169/internalmedicine.49.3525. Epub 2010 Aug 2.
McArdle disease is a glycogenetic myopathy caused by a deficit of myophosphorylase inherited in an autosomal recessive pattern. Here, we report a case of McArdle disease in which fatigability was the only subjective complaint. Objective neurological findings were normal except for very mild muscle weakness in limbs and an elevated serum creatine kinase level. Ischemic forearm exercise test showed deficient glycogenolysis. In the muscle biopsy specimen, periodic acid Schiff (PAS) stained subsarcolemmal glycogen was increased and the muscle phosphorylase A activity was decreased. After administration of vitamin B6, fatigability was diminished and ischemic forearm exercise test showed improved glycogenolysis. Vitamin B6 may be beneficial for McArdle disease, especially for its easy fatigability.
麦克尔迪氏病是一种糖原性肌病,由肌磷酸化酶缺乏引起,呈常染色体隐性遗传模式。在此,我们报告一例麦克尔迪氏病病例,该病例中疲劳是唯一的主观症状。除四肢非常轻微的肌肉无力和血清肌酸激酶水平升高外,客观神经学检查结果正常。缺血性前臂运动试验显示糖原分解不足。在肌肉活检标本中,过碘酸希夫(PAS)染色显示肌膜下糖原增加,肌肉磷酸化酶A活性降低。给予维生素B6后,疲劳减轻,缺血性前臂运动试验显示糖原分解改善。维生素B6可能对麦克尔迪氏病有益,尤其是对其易疲劳症状。