Ikeda Y, Watanabe M, Shoji M
Department of Neurology, Gunma University, School of Medicine.
Nihon Rinsho. 1997 Dec;55(12):3247-52.
Recent genetic research has revealed that hereditary periodic paralysis is an ion channel disorder. Genetic linkage analysis has mapped the autosomal dominant hypokalemic periodic paralysis (HypoPP) locus to chromosome 1q31-32, where the dihydropyridine sensitive calcium channel alpha 1 subunit (CACNL1A3) is located. Subsequently, two predominant missense point mutations were found in the CNCNL1A3 gene. Both mutations substitute arginine to histidine (Arg528His and Arg1239His). The Arg528His mutation is characterized by incomplete penetrance in females, whereas Arg1239His is not. We analyzed Japanese hypokalemic periodic paralysis patients (familial, sporadic and thyrotoxic), and detected the Arg528His mutation in one HypoPP family. This family shows more severe symptoms in successive generations, and the severity of the symptoms is higher in males than in females within the same family.
近期的基因研究表明,遗传性周期性麻痹是一种离子通道疾病。基因连锁分析已将常染色体显性低钾性周期性麻痹(HypoPP)基因座定位于1q31 - 32染色体,即二氢吡啶敏感钙通道α1亚基(CACNL1A3)所在位置。随后,在CNCNL1A3基因中发现了两个主要的错义点突变。这两个突变均将精氨酸替换为组氨酸(Arg528His和Arg1239His)。Arg528His突变的特征是在女性中具有不完全外显率,而Arg1239His则没有。我们分析了日本低钾性周期性麻痹患者(家族性、散发性和甲状腺毒症性),并在一个HypoPP家族中检测到了Arg528His突变。这个家族在连续几代中表现出更严重的症状,并且在同一家庭中男性的症状严重程度高于女性。