Amrhein J A, Meyer W J, Jones H W, Migeon C J
Proc Natl Acad Sci U S A. 1976 Mar;73(3):891-4. doi: 10.1073/pnas.73.3.891.
We have studied ten phenotypically similar patients with complete androgen insensitivity. All of the patients tested had significantly elevated serum luteinizing hormone and plasma androgens within or above the normal adult male range. On the basis of specific dihydrotestosterone binding by skin fibroblasts, we identified two subgroups. Six patients from five different families had undetectable dihydrotestosterone binding, while four patients from two families had normal binding activity. Our results indicate that within the clinical syndrome of androgen insensitivity there are at least two distinct genetic variants. These variants may result from allelic mutations of the same X-linked gene specifying the dihydrotestosterone receptor or, alternatively, from mutations of separate genes both being essential for androgen action in responsive cells.
我们研究了10例具有完全雄激素不敏感症状且表型相似的患者。所有接受检测的患者血清黄体生成素及血浆雄激素均显著升高,处于或高于正常成年男性范围。基于皮肤成纤维细胞对双氢睾酮的特异性结合,我们识别出两个亚组。来自五个不同家庭的6例患者无法检测到双氢睾酮结合,而来自两个家庭的4例患者具有正常的结合活性。我们的结果表明,在雄激素不敏感临床综合征中至少存在两种不同的基因变异。这些变异可能源于指定双氢睾酮受体的同一X连锁基因的等位基因突变,或者源于对反应性细胞中雄激素作用必不可少的两个独立基因的突变。