Kirwan Michael, Beswick Richard, Vulliamy Tom, Nathwani Amit C, Walne Amanda J, Casimir Colin, Dokal Inderjeet
Centre for Paediatrics, Institute of Cell and Molecular Science, Barts and The London School of Medicine and Dentistry, Queen Mary University of London, London, UK.
Br J Haematol. 2009 Mar;144(5):771-81. doi: 10.1111/j.1365-2141.2008.07516.x. Epub 2008 Nov 20.
Dyskeratosis congenita (DC) is an inherited multi-system disorder characterised by muco-cutaneous abnormalities, bone marrow failure and a predisposition to malignancy. Bone marrow failure is the principal cause of mortality and is thought to be the result of premature cell death in the haematopoietic compartment because DC cells age prematurely and tend to have short telomeres. DC is genetically heterogeneous and patients have mutations in genes that encode components of the telomerase complex (DKC1, TERC, TERT, NOP10 and NHP2), and telomere shelterin complex (TINF2), both important in telomere maintenance. Here, we transduced primary T lymphocytes and B lymphocyte lines established from patients with TERC and DKC1 mutations with wild type TERC-bearing lentiviral vectors. We found that transduction with exogenous TERC alone was capable of increasing telomerase activity in mutant T lymphocytes and B lymphocyte lines and improved the survival and thus overall growth of B-lymphocyte lines over a prolonged period, regardless of their disease mutation. Telomeres in TERC-treated lines were longer than in the untreated cultures. This is the first study of its kind in DC lymphocytes and the first to demonstrate that transduction with TERC alone can improve cell survival and telomere length without the need for exogenous TERT.
先天性角化不良(DC)是一种遗传性多系统疾病,其特征为黏膜皮肤异常、骨髓衰竭以及易患恶性肿瘤。骨髓衰竭是主要的死亡原因,被认为是造血细胞过早死亡的结果,因为DC细胞过早衰老且端粒往往较短。DC具有遗传异质性,患者在编码端粒酶复合体(DKC1、TERC、TERT、NOP10和NHP2)和端粒保护蛋白复合体(TINF2)成分的基因中存在突变,这两者在端粒维持中都很重要。在此,我们用携带野生型TERC的慢病毒载体转导从携带TERC和DKC1突变的患者中建立的原代T淋巴细胞和B淋巴细胞系。我们发现,单独用外源性TERC转导能够增加突变T淋巴细胞和B淋巴细胞系中的端粒酶活性,并在较长时间内改善B淋巴细胞系的存活及整体生长,无论其疾病突变情况如何。经TERC处理的细胞系中的端粒比未处理的培养物中的端粒更长。这是首次针对DC淋巴细胞进行此类研究,也是首次证明单独用TERC转导无需外源性TERT就能提高细胞存活率和端粒长度。