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脱支酶缺乏症中的神经肌肉疾病临床类型

Clinical varieties of neuromuscular disease in debrancher deficiency.

作者信息

Cornelio F, Bresolin N, Singer P A, DiMauro S, Rowland L P

出版信息

Arch Neurol. 1984 Oct;41(10):1027-32. doi: 10.1001/archneur.1984.04050210025008.

Abstract

Two men and one woman with debrancher deficiency had symptoms and signs of neuromuscular disease. The two men had adult-onset and slowly progressive weakness, distal muscle wasting, "mixed" electromyographic patterns, and slow nerve conduction velocities; the initial diagnosis was Charcot-Marie-Tooth disease in one patient and motor neuron disease in the other. The woman had stunted growth, delayed motor milestones, and lifelong nonprogressive weakness. A muscle biopsy specimen showed severe vacuolar myopathy in all three cases. The glycogen concentration was increased threefold to sixfold and had an abnormal iodine spectrum. Anaerobic glycolysis in vitro showed impaired use of endogenous and exogenous glycogen but normal use of hexose-phosphate glycolytic intermediates. These three cases illustrated the clinical variety of neuromuscular disease in debrancher deficiency. In patients with weakness of adult onset, the diagnosis is impossible to make without performing a muscle biopsy.

摘要

两名患有脱支酶缺乏症的男性和一名患有该疾病的女性出现了神经肌肉疾病的症状和体征。两名男性发病于成年期,肌无力呈缓慢进展,伴有远端肌肉萎缩、“混合性”肌电图模式以及神经传导速度缓慢;最初,一名患者被诊断为夏科-马里-图斯病,另一名患者被诊断为运动神经元病。该女性生长发育迟缓,运动发育里程碑延迟,且肌无力症状终身无进展。肌肉活检标本显示,所有三例患者均有严重的空泡性肌病。糖原浓度增加了三倍至六倍,且碘光谱异常。体外无氧糖酵解显示,内源性和外源性糖原的利用受损,但己糖磷酸糖酵解中间产物的利用正常。这三例病例说明了脱支酶缺乏症患者神经肌肉疾病的临床多样性。对于成年期出现肌无力症状的患者,不进行肌肉活检就无法做出诊断。

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