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类固醇硫酸酯酶缺乏症

Steroid sulphatase deficiency disease.

作者信息

Lykkesfeldt G, Høyer H, Ibsen H H, Brandrup F

出版信息

Clin Genet. 1985 Sep;28(3):231-7. doi: 10.1111/j.1399-0004.1985.tb00391.x.

Abstract

Seventy-six ichthyotic male patients with a biochemically confirmed diagnosis of steroid sulphatase deficiency are reported. Ascertainment was based on either a previous diagnosis of placental steroid sulphatase deficiency (21 probands and 15 secondary cases), or ichthyosis with steroid sulphatase deficiency (29 probands and 11 secondary cases). The ichthyotic phenotype of the first group was indistinguishable from that of the other group, and completely fitting the classic description of recessive X-linked ichthyosis. A prominent skin peeling in early infancy was found to be a characteristic feature of this syndrome. Maldescent of the testis was registered in 9 patients; and testis cancer had been diagnosed in 2 males with normally descended gonads. This high proportion of patients with gonadal abnormalities strongly indicates a relation with the steroid sulphatase deficiency. Corneal opacities, not affecting visual acuity, were seen in 14 out of 28 males by slit-lamp examination.

摘要

报告了76例经生化确诊为类固醇硫酸酯酶缺乏症的鱼鳞病男性患者。确诊依据为既往胎盘类固醇硫酸酯酶缺乏症诊断(21例先证者和15例继发病例),或鱼鳞病合并类固醇硫酸酯酶缺乏症(29例先证者和11例继发病例)。第一组鱼鳞病表型与另一组无差异,完全符合隐性X连锁鱼鳞病的经典描述。婴儿早期明显的皮肤脱屑是该综合征的一个特征性表现。9例患者存在睾丸下降不全;2例性腺正常的男性被诊断为睾丸癌。性腺异常患者比例如此之高,强烈表明与类固醇硫酸酯酶缺乏症有关。裂隙灯检查发现,28例男性中有14例存在不影响视力的角膜混浊。

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