Vulliamy Tom, Marrone Anna, Dokal Inderjeet, Mason Philip J
Department of Haematology, Division of Investigative Science, Faculty of Medicine, Imperial College of Science, Technology and Medicine, Hammersmith Hospital, London W12 0NN, UK.
Lancet. 2002 Jun 22;359(9324):2168-70. doi: 10.1016/S0140-6736(02)09087-6.
The main cause of aplastic anaemia remains elusive. Germline mutations in the gene encoding the RNA component of telomerase (hTR) have been seen in the autosomal dominant form of dyskeratosis congenita--an inherited syndrome characterised by aplastic anaemia. By screening the hTR gene, we identified mutations in two of 17 patients with idiopathic aplastic anaemia, three of 27 patients with constitutional aplastic anaemia, but in none of 214 normal controls (p<0.0001). Furthermore, patients with hTR mutations had significantly shorter telomeres than age-matched controls (p=0.027). These data indicate that, in a subset of patients with aplastic anaemia, the disorder might be associated with a genetic lesion in the telomere maintenance pathway.
再生障碍性贫血的主要病因仍不清楚。在常染色体显性遗传的先天性角化不良(一种以再生障碍性贫血为特征的遗传性综合征)中,已发现编码端粒酶RNA成分(hTR)的基因存在种系突变。通过筛查hTR基因,我们在17例特发性再生障碍性贫血患者中的2例、27例体质性再生障碍性贫血患者中的3例发现了突变,但在214例正常对照中均未发现突变(p<0.0001)。此外,hTR突变患者的端粒明显短于年龄匹配的对照(p=0.027)。这些数据表明,在一部分再生障碍性贫血患者中,该疾病可能与端粒维持途径中的遗传损伤有关。