Chan K-W, Chen T, Jiang L, Fok S F-S, Lee T-L, Lee B-W, Yang X, Lau Y-L
Department of Paediatrics and Adolescent Medicine, The University of Hong Kong.
Int J Immunogenet. 2006 Jun;33(3):205-9. doi: 10.1111/j.1744-313X.2006.00598.x.
X-linked agammaglobulinaemia (XLA) is an immunodeficiency caused by Bruton tyrosine kinase (BTK) gene mutations. The disease is characterized by recurrent bacterial infections and profound hypogammaglobulinemia with marked reduction or lack of mature B-cells in the peripheral blood. Molecular characterization of BTK gene provides an opportunity for definitive diagnosis of XLA patients, especially for those with atypical phenotype resulting in a milder or late-onset form of the disease. The diagnosis allows accurate carrier detection with subsequent genetic counselling and prenatal diagnosis. In this study, long polymerase chain reaction (PCR)-direct sequencing analysis of the BTK gene in 12 unrelated Chinese XLA patients had been performed. Eight recurrent mutations and four novel mutations were identified. This is the first report of Chinese cases from three different East Asia regions together, including Hong Kong, Singapore and mainland China. Future clinical and genetic information from the undiagnosed Chinese XLA patients may provide insight into the genotype-phenotype correlations of BTK gene.
X连锁无丙种球蛋白血症(XLA)是一种由布鲁顿酪氨酸激酶(BTK)基因突变引起的免疫缺陷病。该疾病的特征为反复发生细菌感染、严重低丙种球蛋白血症,外周血中成熟B细胞显著减少或缺乏。BTK基因的分子特征为明确诊断XLA患者提供了机会,尤其是对于那些具有非典型表型、导致疾病症状较轻或发病较晚的患者。该诊断有助于准确检测携带者,随后进行遗传咨询和产前诊断。在本研究中,对12名来自中国不同地区且无亲缘关系的XLA患者进行了BTK基因的长聚合酶链反应(PCR)-直接测序分析。共鉴定出8个复发突变和4个新突变。这是首次将来自中国香港、新加坡和中国大陆这三个不同东亚地区的病例合并在一起进行报道。未来来自未确诊的中国XLA患者的临床和遗传信息可能有助于深入了解BTK基因的基因型-表型相关性。