Aicardi J, Barbosa C, Andermann E, Andermann F, Morcos R, Ghanem Q, Fukuyama Y, Awaya Y, Moe P
INSERM Hôpital des Enfants Malades, Paris, France.
Ann Neurol. 1988 Oct;24(4):497-502. doi: 10.1002/ana.410240404.
We report 14 patients with a slowly progressive syndrome featuring ataxia, choreoathetosis, and ocular motor apraxia in both the horizontal and vertical planes. Although the neurological signs were indistinguishable from those of ataxia-telangiectasia, the onset tended to be later and none of the patients had evidence of multisystemic involvement. Specifically, there was no tendency to frequent infections, and immunoglobulins, alpha-fetoprotein, T- and B-lymphocyte markers, and chromosomes 7 and 14 were normal in all tested patients. The simultaneous absence of telangiectasias and of other nonneurological manifestations made ataxia-telangiectasia an unlikely diagnosis. We suggest that these patients suffer from an unusual type of spinocerebellar degeneration. This syndrome has been observed in different populations from three continents, with a genetic pattern suggesting recessive autosomal inheritance.
我们报告了14例患有缓慢进展综合征的患者,其特征为共济失调、舞蹈手足徐动症以及水平和垂直平面的眼球运动失用症。尽管这些神经体征与共济失调毛细血管扩张症难以区分,但发病往往较晚,且所有患者均无多系统受累的证据。具体而言,没有频繁感染的倾向,所有受试患者的免疫球蛋白、甲胎蛋白、T和B淋巴细胞标志物以及7号和14号染色体均正常。同时不存在毛细血管扩张和其他非神经学表现,使得共济失调毛细血管扩张症不太可能被诊断。我们认为这些患者患有一种不寻常类型的脊髓小脑变性。该综合征在来自三大洲的不同人群中均有观察到,其遗传模式提示为隐性常染色体遗传。