Sulewski J M, Ward S, Ladda R L
J Med Genet. 1980 Aug;17(4):321-3. doi: 10.1136/jmg.17.4.321.
The proband was evaluated at 19 years of age because of primary amenorrhoea and, on chromosomal analysis, was found to have a 46,XY karyotype in 75% of her cells and 48,XY, +8, +21 in 25% of her cells. She appeared normal at birth and exhibited normal intellectual and physical development until puberty when secondary sexual differentiation failed. This young women showed none of the dysmorphic features associated with either trisomy 8 or trisomy 21. Her XY gonadal dysgenesis was manifested by late developmental problems of amenorrhoea, sexual infantilism, and gonadal neoplasia.
先证者因原发性闭经于19岁时接受评估,染色体分析发现其75%的细胞具有46,XY核型,25%的细胞具有48,XY, +8, +21核型。她出生时外观正常,在青春期继发性性分化失败之前,智力和身体发育均正常。这位年轻女性没有表现出与8号三体或21号三体相关的畸形特征。她的XY性腺发育不全表现为闭经、性幼稚和性腺肿瘤等发育后期问题。