Loughran T P, Hammond W P
J Exp Med. 1986 Dec 1;164(6):2089-94. doi: 10.1084/jem.164.6.2089.
Human cyclic neutropenia occurs in children and adults. Adult-onset cyclic neutropenia is an acquired disease characterized by increased numbers of large granular lymphocytes (LGL), in contrast to childhood-onset cyclic neutropenia in which LGL counts are normal. We investigated the clonality of lymphocytes in these two groups of patients by assessing the rearrangement status of the T cell receptor beta chain gene. Patients with adult-onset cyclic neutropenia showed clonal rearrangement of the T beta gene whereas the children did not. Since LGL are known to have multiple regulatory effects on normal hematopoiesis, the finding of a clonal proliferation of this lymphocyte population implicates these cells in the pathogenesis of cyclic neutropenia.
人类周期性中性粒细胞减少症发生于儿童和成人。成人起病的周期性中性粒细胞减少症是一种获得性疾病,其特征是大颗粒淋巴细胞(LGL)数量增加,这与儿童起病的周期性中性粒细胞减少症不同,后者的LGL计数正常。我们通过评估T细胞受体β链基因的重排状态,研究了这两组患者淋巴细胞的克隆性。成人起病的周期性中性粒细胞减少症患者显示Tβ基因发生克隆性重排,而儿童患者则未出现。由于已知LGL对正常造血有多种调节作用,这一淋巴细胞群体的克隆性增殖表明这些细胞参与了周期性中性粒细胞减少症的发病机制。