Igarashi S, Tanno Y, Onodera O, Yamazaki M, Sato S, Ishikawa A, Miyatani N, Nagashima M, Ishikawa Y, Sahashi K
Department of Neurology, Nishi-Ojiya Byoin National Sanatorium, Niigata, Japan.
Neurology. 1992 Dec;42(12):2300-2. doi: 10.1212/wnl.42.12.2300.
X-linked spinal and bulbar muscular atrophy (SBMA), a motor neuron disease associated with androgen insensitivity, is caused by androgen receptor gene mutations with an increased number of tandem CAG repeats in exon 1. We investigated the increased number of CAG repeats in androgen receptor genes of 19 SBMA patients and found that this correlated strongly with the age at onset of muscle weakness. Thus, SBMA is the first genetic disease in which a strong correlation between the degree of genetic abnormality (number of CAG tandem repeats) and clinical phenotypic expression is demonstrable. The results further indicate that androgen gene mutation is directly involved in the degeneration of motor neurons.
X连锁脊髓和延髓性肌萎缩症(SBMA)是一种与雄激素不敏感相关的运动神经元疾病,由雄激素受体基因突变引起,该基因外显子1中的串联CAG重复序列数量增加。我们研究了19例SBMA患者雄激素受体基因中CAG重复序列数量的增加情况,发现这与肌肉无力的发病年龄密切相关。因此,SBMA是第一种可证明遗传异常程度(CAG串联重复序列数量)与临床表型表达之间存在强相关性的遗传疾病。结果还进一步表明,雄激素基因突变直接参与运动神经元的退化。