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大剂量硫胺素可改善弗里德赖希共济失调的症状。

High-dose thiamine improves the symptoms of Friedreich's ataxia.

作者信息

Costantini Antonio, Giorgi Rafaela, D'Agostino Sonia, Pala Maria Immacolata

机构信息

Department of Neurological Rehabilitation, Villa Immacolata, Viterbo, Italy.

出版信息

BMJ Case Rep. 2013 May 22;2013:bcr2013009424. doi: 10.1136/bcr-2013-009424.

Abstract

Friedreich's ataxia (FRDA) is an autosomal recessive inherited disorder characterised by progressive gait and limb ataxia, dysarthria, areflexia, loss of position sense and a progressive motor weakness of central origin. Some observations indicate that all symptoms of FRDA ataxia could be the manifestation of a thiamine deficiency because of enzymatic abnormalities. Two patients with FRDA were under rehabilitative treatment from February 2012 to February 2013. The scale for assessment and rating of ataxia was performed. The patient began an intramuscular therapy with 100 mg of thiamine every 3-5 days. Injection of high-dose thiamine was effective in reversing the motor failure. From this clinical observation, it is reasonable to infer that a thiamine deficiency due to enzymatic abnormalities could cause a selective neuronal damage in the centres that are typically affected by this disease.

摘要

弗里德赖希共济失调(FRDA)是一种常染色体隐性遗传性疾病,其特征为进行性步态和肢体共济失调、构音障碍、反射消失、位置觉丧失以及中枢性进行性运动无力。一些观察结果表明,由于酶异常,FRDA共济失调的所有症状可能是硫胺素缺乏的表现。2012年2月至2013年2月,两名FRDA患者接受了康复治疗。进行了共济失调评估和评级量表。患者开始每3 - 5天接受一次100毫克硫胺素的肌肉注射治疗。大剂量硫胺素注射有效逆转了运动功能衰竭。从这一临床观察结果合理推断出,由于酶异常导致的硫胺素缺乏可能会在通常受该疾病影响的中枢引起选择性神经元损伤。

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