Koch M C, Ricker K, Otto M, Grimm T, Bender K, Zoll B, Harper P S, Lehmann-Horn F, Rüdel R, Hoffman E P
Abteilung Allgemeine Humangenetik, Philipps Universität, Marburg, Federal Republic of Germany.
Hum Genet. 1991 Nov;88(1):71-4. doi: 10.1007/BF00204932.
Paramyotonia congenita (PC), an autosomal dominant non-progressive muscle disorder, is characterised by cold-induced stiffness followed by muscle weakness. The weakness is caused by a dysfunction of the sodium channel in muscle fibre. Parts of the gene coding for the alpha-subunit of the sodium channel of the adult human skeletal muscle (SCN4A) have been localised on chromosome 17. To investigate the role of this gene in the etiology of PC, a linkage analysis in 17 well-defined families was carried out. The results (zeta = 20.61, theta = 0.001) show that the mutant gene responsible for the disorder is indeed tightly linked to the SCN4A gene. The mutation causing hyperkalemic periodic paralysis (HyperPP) with myotonia has previously been mapped to this gene locus by the same candidate gene approach. Thus, our data suggest that PC and HyperPP are caused by allelic mutations at a single locus on chromosome 17.
先天性副肌强直(PC)是一种常染色体显性非进行性肌肉疾病,其特征为冷诱导的僵硬,随后出现肌肉无力。这种无力是由肌纤维中钠通道功能障碍引起的。编码成人体骨骼肌钠通道α亚基的部分基因(SCN4A)已定位在17号染色体上。为了研究该基因在PC病因学中的作用,对17个明确界定的家族进行了连锁分析。结果(ζ = 20.61,θ = 0.001)表明,导致该疾病的突变基因确实与SCN4A基因紧密连锁。先前通过相同的候选基因方法已将导致伴有肌强直的高钾性周期性麻痹(HyperPP)的突变定位到该基因座。因此,我们的数据表明,PC和HyperPP是由17号染色体上单个位点的等位基因突变引起的。