Brown P, Cervenáková L, Goldfarb L G, McCombie W R, Rubenstein R, Will R G, Pocchiari M, Martinez-Lage J F, Scalici C, Masullo C
Laboratory of CNS Studies, National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD 20892.
Neurology. 1994 Feb;44(2):291-3. doi: 10.1212/wnl.44.2.291.
We tested DNA from 15 centrally infected cases of iatrogenic Creutzfeldt-Jakob disease (CJD) (dura mater or corneal homografts and stereotactic EEG electrodes), 11 peripherally infected cases (native human growth hormone or gonadotrophin), and 110 control individuals for the presence of mutations in the chromosome 20 amyloid gene. No patient or control had any of the known pathogenic point or insert mutations found in familial disease, but allelic homozygosity at polymorphic codon 129 was present in all but two (92%) of the 26 patients, compared with 54 (50%) of the 110 controls (p < 0.001). Pooled data from all identified and tested cases of iatrogenic disease yielded a worldwide total of 56 patients, of whom all but four were homozygous at codon 129 (p < 0.001). These findings support the thesis that homozygosity at codon 129 enhances susceptibility to iatrogenic infections of both central and peripheral origin, with evident implications for the population of dura mater homograft and pituitary hormone recipients whose lives have been complicated by the possibility of exposure to the infectious agent of CJD.
我们检测了15例医源性克雅氏病(CJD)中枢感染病例(硬脑膜或角膜同种异体移植以及立体定向脑电图电极)、11例外周感染病例(天然人生长激素或促性腺激素)以及110名对照个体的20号染色体淀粉样蛋白基因突变情况。患者和对照中均未发现家族性疾病中已知的致病点突变或插入突变,但26例患者中有24例(92%)在多态密码子129处存在等位基因纯合性,而110名对照中有54例(50%)存在该情况(p<0.001)。所有已识别和检测的医源性疾病病例的汇总数据显示,全球共有56例患者,其中除4例外在密码子129处均为纯合子(p<0.001)。这些发现支持了这样一种观点,即密码子129处的纯合性会增加中枢和外周源性医源性感染的易感性,这对硬脑膜同种异体移植受者和垂体激素接受者群体具有明显影响,他们的生活因可能接触CJD感染因子而变得复杂。