Engelhard D, Landreth K S, Kapoor N, Kincade P W, De Bault L E, Theodore A, Good R A
Proc Natl Acad Sci U S A. 1983 Sep;80(18):5734-8. doi: 10.1073/pnas.80.18.5734.
A 16-month-old male patient with cyclic neutropenia was found to have cyclic fluctuations of monocytes, lymphocytes, platelets, and eosinophils in the peripheral blood. Changes in lymphocyte counts were not obviously related to B, T, or natural killer cells. All classes of immunoglobulins were elevated throughout the cycle. Studies of the marrow morphology revealed remarkable cyclic oscillations of lymphoid as well as myeloid lineage cells. Granulocyte-macrophage progenitors (CFU-c) cycled and were virtually absent 1 wk prior to the neutropenic nadir. The cyclic changes in marrow lymphoid cell numbers were primarily due to changes in numbers of surface immunoglobulin negative (sIg-), cytoplasmic Ig+ (cIg+) pre-B cells. Pre-B cell numbers cycled from normal to extraordinarily elevated values with the same periodicity but reciprocal to the neutrophil cycle. We propose that the primary defect in cyclic neutropenia may either be a periodic failure of an early myeloid differentiation factor or a blunted response of early myeloid precursors to a common hemopoietic growth factor. This may lead to periodic fluctuations in the production or delivery of growth factors (or factor) that influence early stages of differentiation of other hemopoietic cells, including pre-B cells. The essential periodic deficiency is consequently reflected in deficient production of CFU-c accompanied by excessive production or accumulation of pre-B cells (and probably other hemopoietic precursors) in the marrow.
一名患有周期性中性粒细胞减少症的16个月大男性患者,其外周血中的单核细胞、淋巴细胞、血小板和嗜酸性粒细胞出现周期性波动。淋巴细胞计数的变化与B细胞、T细胞或自然杀伤细胞无明显关联。在整个周期中,各类免疫球蛋白均升高。骨髓形态学研究显示,淋巴系和髓系细胞均有显著的周期性振荡。粒细胞-巨噬细胞祖细胞(CFU-c)呈周期性变化,在中性粒细胞减少最低点前1周几乎消失。骨髓淋巴样细胞数量的周期性变化主要归因于表面免疫球蛋白阴性(sIg-)、细胞质Ig+(cIg+)前B细胞数量的变化。前B细胞数量从正常水平周期性地异常升高,其周期相同但与中性粒细胞周期相反。我们认为,周期性中性粒细胞减少症的主要缺陷可能是早期髓系分化因子的周期性功能障碍,或者是早期髓系前体细胞对常见造血生长因子的反应迟钝。这可能导致影响其他造血细胞(包括前B细胞)早期分化阶段的生长因子(或多种因子)的产生或释放出现周期性波动。因此,基本的周期性缺陷表现为CFU-c生成不足,同时骨髓中前B细胞(可能还有其他造血前体细胞)过度生成或积聚。