Liu Wei, Narayanan Vinodh
Department of Neurology, Children's Health Center, St. Joseph's Hospital and Medical Center, Phoenix, AZ 85013, USA.
Semin Pediatr Neurol. 2008 Dec;15(4):216-20. doi: 10.1016/j.spen.2008.10.014.
Ataxia-telangiectasia (AT) belongs to a group of recessively inherited disorders characterized by progressive ataxia and oculomotor apraxia. Included in this group are AT, ataxia-telangiectasia-like disorder (ATLD), ataxia with oculomotor apraxia type 1 (AOA 1), ataxia with oculomotor apraxia type 2 (AOA 2), and the recently described AOA3. Common to this group is the underlying cellular defect in the recognition and repair of double-strand or single-strand DNA breaks. Clinical and laboratory features allow one to distinguish between these various disorders. In this report, we describe a child with early onset progressive ataxia, oculomotor apraxia, ocular telangiectasia, and white-matter changes by magnetic resonance imaging, which appears to be yet another novel form of AOA. We designate this condition as AOA-WM to call attention to the central demyelination seen in this variety of ataxia with oculomotor apraxia.
共济失调毛细血管扩张症(AT)属于一组隐性遗传性疾病,其特征为进行性共济失调和眼球运动失用症。这组疾病包括AT、共济失调毛细血管扩张症样疾病(ATLD)、1型眼球运动失用症伴共济失调(AOA 1)、2型眼球运动失用症伴共济失调(AOA 2)以及最近描述的AOA3。这组疾病的共同特点是在双链或单链DNA断裂的识别和修复方面存在潜在的细胞缺陷。临床和实验室特征有助于区分这些不同的疾病。在本报告中,我们描述了一名儿童,其早期出现进行性共济失调、眼球运动失用症、眼部毛细血管扩张症,且磁共振成像显示有白质改变,这似乎是AOA的另一种新形式。我们将这种病症命名为AOA-WM,以引起对这种伴有眼球运动失用症的共济失调中所见中枢性脱髓鞘的关注。