Wong K S, Lai C, Ng H K
Department of Medicine, Chinese University of Hong Kong, Prince of Wales Hospital, Shatin.
Clin Exp Neurol. 1991;28:210-8.
Late-onset acid maltase deficiency is a rare disorder. We describe a nineteen year old Chinese girl who presented with diarrhoea, limb-girdle weakness and respiratory failure requiring mechanical ventilation. Electromyography showed polyphasic potentials and myotonic discharges. Muscle biopsy revealed features characteristic of acid maltase deficiency. Assay of acid alpha-glucosidase in cultured skin fibroblasts confirmed the diagnosis. Supportive treatment with nocturnal intermittent positive pressure ventilation via a nasal mask and dietary supplementation with branched-chain aminoacids proved effective in this patient. The cause of diarrhoea remained uncertain. This is the first documented case of acid maltase deficiency in Chinese adult.
晚发型酸性麦芽糖酶缺乏症是一种罕见的疾病。我们描述了一名19岁的中国女孩,她出现腹泻、肢带肌无力和呼吸衰竭,需要机械通气。肌电图显示多相电位和肌强直放电。肌肉活检显示出酸性麦芽糖酶缺乏症的特征性表现。对培养的皮肤成纤维细胞中的酸性α-葡萄糖苷酶进行检测证实了诊断。通过鼻罩进行夜间间歇正压通气的支持性治疗以及补充支链氨基酸的饮食疗法在该患者中被证明是有效的。腹泻的原因仍不确定。这是中国成年人中首例有记录的酸性麦芽糖酶缺乏症病例。