Ballabio A, Sebastio G, Carrozzo R, Parenti G, Piccirillo A, Persico M G, Andria G
Department of Paediatrics, Second Faculty of Medicine, University of Naples, Italy.
Hum Genet. 1987 Dec;77(4):338-41. doi: 10.1007/BF00291422.
We have studied 16 men, from 10 unrelated Italian families, affected by steroid suphatase (STS) deficiency, which is the basic defect of X-linked ichthyosis (XLI). The patients' clinical diagnoses were of either isolated ichthyosis or ichthyosis associated with Kallmann syndrome (KS) (hypogonadotropic hypogonadism and anosmia). DNA from patients and their relatives was analysed by Southern blotting followed by hydridization with an STS cDNA probe. None of the patients affected by either XLI or XLI/KS showed any hybridization signal, thus revealing a deletion in the STS gene. We suggest that a gene deletion may be the most common molecular defect involved in XLI and that the syndrome XLI/KS may be due to a deletion of both the STS and the KS loci.
我们研究了来自10个无亲缘关系的意大利家庭的16名男性,他们患有类固醇硫酸酯酶(STS)缺乏症,这是X连锁鱼鳞病(XLI)的基本缺陷。患者的临床诊断为单纯性鱼鳞病或与卡尔曼综合征(KS)(促性腺激素缺乏性性腺功能减退和嗅觉缺失)相关的鱼鳞病。通过Southern印迹法分析患者及其亲属的DNA,然后用STS cDNA探针进行杂交。受XLI或XLI/KS影响的患者均未显示任何杂交信号,从而揭示了STS基因的缺失。我们认为基因缺失可能是XLI中最常见的分子缺陷,并且综合征XLI/KS可能是由于STS和KS基因座均缺失所致。