Villa A, Notarangelo L, Macchi P, Mantuano E, Cavagni G, Brugnoni D, Strina D, Patrosso M C, Ramenghi U, Sacco M G
Istituto di Tecnologie Biomediche Avanzate, CNR, Milano, Italy.
Nat Genet. 1995 Apr;9(4):414-7. doi: 10.1038/ng0495-414.
X-linked thrombocytopenia (XLT) is a rare recessive hereditary disorder characterized by isolated thrombocytopenia with small-sized platelets. The XLT locus has been located to chromosome Xp11 by linkage analysis, which is also where the recently cloned Wiskott-Aldrich syndrome (WAS) gene, maps. The relationship between XLT and WAS has long been debated; they might be due to different mutations of the same gene or to mutations in different genes. We now show that mutations in the WAS gene, different from those found in WAS patients, are present in three unrelated male patients with isolated thrombocytopenia and small-sized platelets. Our results demonstrate that XLT and WAS are allelic forms of the same disease, but the causes of the differences need to be further investigated.
X连锁血小板减少症(XLT)是一种罕见的隐性遗传性疾病,其特征为单纯性血小板减少伴小血小板。通过连锁分析已将XLT基因座定位于Xp11染色体,而最近克隆的威斯科特-奥尔德里奇综合征(WAS)基因也定位于此。XLT与WAS之间的关系长期以来一直存在争议;它们可能是同一基因的不同突变所致,也可能是不同基因的突变所致。我们现在发现,在三名患有单纯性血小板减少和小血小板的无关男性患者中存在与WAS患者不同的WAS基因突变。我们的结果表明,XLT和WAS是同一种疾病的等位基因形式,但差异的原因有待进一步研究。