Wada Takahito, Kobayashi Norio, Takahashi Yoshio, Aoki Tomoko, Watanabe Takako, Saitoh Shinji
Department of Pediatrics, Hokkaido University School of Medicine, N-15 W-7, Kita-ku, Sapporo 060-8638, Japan.
Pediatr Neurol. 2002 Jan;26(1):47-50. doi: 10.1016/s0887-8994(01)00371-x.
We report a Japanese family carrying a T666M missense mutation of CACNA1A. Affected members demonstrated a strikingly wide clinical spectrum including migraine, hemiplegia, coma, and progressive cerebellar ataxia. Despite such variability of the clinical features, they demonstrated similar magnetic resonance imaging findings demonstrating cerebellar atrophy predominantly of the cerebellar vermis. These magnetic resonance images appeared not to correlate with clinical severity. Our findings should indicate that a T666M mutation of CACNA1A may be associated with more variable clinical features and that paroxysmal hemiplegic migraine attacks and progressive cerebellar atrophy should have distinct mechanisms of pathogenesis.
我们报告了一个携带CACNA1A基因T666M错义突变的日裔家族。受影响的成员表现出极为广泛的临床谱,包括偏头痛、偏瘫、昏迷和进行性小脑共济失调。尽管临床特征存在这种变异性,但他们的磁共振成像结果相似,均显示主要为小脑蚓部萎缩。这些磁共振图像似乎与临床严重程度无关。我们的研究结果表明,CACNA1A基因的T666M突变可能与更多变的临床特征相关,并且阵发性偏瘫性偏头痛发作和进行性小脑萎缩应有不同的发病机制。