Bagley Jessamyn, Cortes Maria L, Breakefield Xandra O, Iacomini John
Transplantation Biology Research Center, Massachusetts General Hospital, MGH-East, 149-5210 13th St, Boston, MA 02129, USA.
Blood. 2004 Jul 15;104(2):572-8. doi: 10.1182/blood-2003-12-4226. Epub 2004 Mar 25.
Ataxia-telangiectasia (A-T) is a human autosomal recessive disease caused by mutations in the gene encoding ataxia-telangiectasia mutated (ATM). A-T is characterized by progressive cerebellar degeneration, variable immunodeficiency, and a high incidence of leukemia and lymphoma. Recurrent sino-pulmonary infections secondary to immunodeficiency and hematopoietic malignancies are major causes of morbidity and mortality in A-T patients. In mice, an introduced mutation in Atm leads to a phenotype that recapitulates many of the symptoms of A-T, including immune system abnormalities and susceptibility to malignancy. Here we show that the replacement of the bone marrow compartment in Atm knockout mice (Atm(-/-)) using a clinically relevant, nonmyeloablative host-conditioning regimen can be used to overcome the immune deficiencies and prevent the malignancies observed in these mice. Therefore, bone marrow transplantation may prove to be of therapeutic benefit in A-T patients.
共济失调毛细血管扩张症(A-T)是一种人类常染色体隐性疾病,由编码共济失调毛细血管扩张症突变基因(ATM)的基因突变引起。A-T的特征是进行性小脑变性、可变免疫缺陷以及白血病和淋巴瘤的高发病率。免疫缺陷和造血系统恶性肿瘤继发的反复肺部感染是A-T患者发病和死亡的主要原因。在小鼠中,Atm基因的导入突变会导致一种表型,该表型概括了A-T的许多症状,包括免疫系统异常和对恶性肿瘤的易感性。在这里,我们表明,使用临床相关的非清髓性宿主预处理方案替换Atm基因敲除小鼠(Atm(-/-))的骨髓区室,可用于克服这些小鼠中观察到的免疫缺陷并预防恶性肿瘤。因此,骨髓移植可能对A-T患者具有治疗益处。