Amrhein J A, Klingensmith G J, Walsh P C, McKusick V A, Migeon C J
N Engl J Med. 1977 Aug 18;297(7):350-6. doi: 10.1056/NEJM197708182970703.
We investigated eight patients with the Reifenstein syndrome to define the hormonal basis for this condition. The patients had normal or elevated concentrations of plasma androgens, normal production rates of testosterone and dihydrotestosterone, elevated serum levels of luteinizing hormone and normal 5alpha-reductase activity in skin fibroblasts. These findings indicate that the syndrome results from defective androgen action rather than from decreased androgen synthesis. The term "partial androgen insensitivity syndrome" describes this condition more accurately than a term based on clinical phenotype. Dihydrotestosterone binding studies in skin fibroblasts demonstrated two genetic variants similar to those reported in complete androgen insensitivity syndrome. One patient had a partial deficiency of cytoplasmic dihydrotestosterone binding, and four others had normal binding activity. The cause of the androgen insensitivity in the last four cases is unknown. Treatment with testosterone suppressed serum luteinizing hormone levels and promoted mild virilizing effects.
我们研究了8例患有赖芬斯坦综合征的患者,以确定该病症的激素基础。这些患者的血浆雄激素浓度正常或升高,睾酮和双氢睾酮的生成率正常,促黄体生成素血清水平升高,皮肤成纤维细胞中的5α-还原酶活性正常。这些发现表明,该综合征是由雄激素作用缺陷引起的,而非雄激素合成减少所致。“部分雄激素不敏感综合征”这一术语比基于临床表型的术语更准确地描述了这种情况。皮肤成纤维细胞中的双氢睾酮结合研究显示出两种与完全雄激素不敏感综合征中所报道的类似的基因变异。1例患者存在细胞质双氢睾酮结合部分缺陷,另外4例患者的结合活性正常。后4例患者雄激素不敏感的原因尚不清楚。睾酮治疗可抑制血清促黄体生成素水平并产生轻度的男性化作用。