Crow Y J, Jackson A P, Roberts E, van Beusekom E, Barth P, Corry P, Ferrie C D, Hamel B C, Jayatunga R, Karbani G, Kálmánchey R, Kelemen A, King M, Kumar R, Livingstone J, Massey R, McWilliam R, Meager A, Rittey C, Stephenson J B, Tolmie J L, Verrips A, Voit T, van Bokhoven H, Brunner H G, Woods C G
Molecular Medicine Unit, Clinical Sciences Building, St. James's University Hospital, Leeds, United Kingdom.
Am J Hum Genet. 2000 Jul;67(1):213-21. doi: 10.1086/302955. Epub 2000 May 25.
We have studied 23 children from 13 families with a clinical diagnosis of Aicardi-Goutières syndrome. Affected individuals had developed an early-onset progressive encephalopathy that was characterized by a normal head circumference at birth, basal ganglia calcification, negative viral studies, and abnormalities of cerebrospinal fluid comprising either raised white cell counts and/or raised levels of interferon-alpha. By means of genomewide linkage analysis, a maximum-heterogeneity LOD score of 5.28 was reached at marker D3S3563, with alpha=.48, where alpha is the proportion of families showing linkage. Our data suggest the existence of locus heterogeneity in Aicardi-Goutières syndrome and highlight potential difficulties in the differentiation of this condition from pseudo-TORCH (toxoplasmosis, rubella, cytomegalovirus, and herpes simplex virus types 1 and 2) syndrome.
我们研究了来自13个家庭的23名临床诊断为艾卡迪-古铁雷斯综合征的儿童。受影响个体出现了早发性进行性脑病,其特征为出生时头围正常、基底节钙化、病毒学检查阴性,以及脑脊液异常,包括白细胞计数升高和/或α-干扰素水平升高。通过全基因组连锁分析,在标记D3S3563处达到了最大异质性LOD分数5.28,α = 0.48,其中α是显示连锁的家庭比例。我们的数据表明艾卡迪-古铁雷斯综合征存在基因座异质性,并突出了将这种疾病与假TORCH(弓形虫病、风疹、巨细胞病毒以及1型和2型单纯疱疹病毒)综合征区分开来的潜在困难。