Literature DB >> 23704441

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

Antonio Costantini1, Rafaela Giorgi, Sonia D'Agostino, Maria Immacolata Pala.   

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.

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Year:  2013        PMID: 23704441      PMCID: PMC3669970          DOI: 10.1136/bcr-2013-009424

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  14 in total

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6.  Electrocardiogram in Friedreich's ataxia: A short-term surrogate endpoint for treatment efficacy.

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