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1型动眼神经失用性共济失调(AOA1):2例新患者的临床和神经心理学特征及鉴别诊断

Ataxia with oculomotor apraxia type 1 (AOA1): clinical and neuropsychological features in 2 new patients and differential diagnosis.

作者信息

D'Arrigo Stefano, Riva Daria, Bulgheroni Sara, Chiapparini Luisa, Castellotti Barbara, Gellera Cinzia, Pantaleoni Chiara

机构信息

Developmental Neurology Department, Istituto Neurologico C. Besta, Milan, Italy.

出版信息

J Child Neurol. 2008 Aug;23(8):895-900. doi: 10.1177/0883073808314959. Epub 2008 Apr 10.

Abstract

Ataxia with oculomotor apraxia type 1 (AOA1) is an autosomal recessive disease characterized by early-onset and slowly progressive cerebellar ataxia, areflexia, and peripheral neuropathy. Ocular apraxia is most prominent in the early stage of the disease, by contrast, hypoalbuminemia, hypercholesterolemia, and cognitive impairment are present in the adult stage. AOA1 is caused by a mutation in the APTX gene (9p13.3) encoding a nuclear protein named aprataxin, which is involved in the mechanism of DNA repair. We report here the clinical features of 2 patients with mutations in the APTX gene, and we discuss the differential diagnosis with other forms of hereditary ataxia.

摘要

1型伴动眼神经失用性共济失调(AOA1)是一种常染色体隐性疾病,其特征为早发性和缓慢进展性小脑共济失调、无反射和周围神经病变。眼失用在疾病早期最为突出,相比之下,低白蛋白血症、高胆固醇血症和认知障碍则出现在成年期。AOA1由APTX基因(9p13.3)突变引起,该基因编码一种名为共济失调伴动眼神经失用蛋白的核蛋白,其参与DNA修复机制。我们在此报告2例APTX基因突变患者的临床特征,并讨论与其他形式遗传性共济失调的鉴别诊断。

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