Westerberg Lisa S, de la Fuente Miguel A, Wermeling Fredrik, Ochs Hans D, Karlsson Mikael C I, Snapper Scott B, Notarangelo Luigi D
Gastrointestinal Unit and the Center for the Study of Inflammatory Bowel Diseases, Massachusetts General Hospital, Boston, USA.
Blood. 2008 Nov 15;112(10):4139-47. doi: 10.1182/blood-2008-02-140715. Epub 2008 Sep 4.
Development of hematopoietic cells depends on a dynamic actin cytoskeleton. Here we demonstrate that expression of the cytoskeletal regulator WASP, mutated in the Wiskott-Aldrich syndrome, provides selective advantage for the development of naturally occurring regulatory T cells, natural killer T cells, CD4(+) and CD8(+) T lymphocytes, marginal zone (MZ) B cells, MZ macrophages, and platelets. To define the relative contribution of MZ B cells and MZ macrophages for MZ development, we generated wild-type and WASP-deficient bone marrow chimeric mice, with full restoration of the MZ. However, even in the presence of MZ macrophages, only 10% of MZ B cells were of WASP-deficient origin. We show that WASP-deficient MZ B cells hyperproliferate in vivo and fail to respond to sphingosine-1-phosphate, a crucial chemoattractant for MZ B-cell positioning. Abnormalities of the MZ compartment in WASP(-/-) mice lead to aberrant uptake of Staphylococcus aureus and to a reduced immune response to TNP-Ficoll. Moreover, WASP-deficient mice have increased levels of "natural" IgM antibodies. Our findings reveal that WASP regulates both development and function of hematopoietic cells. We demonstrate that WASP deficiency leads to an aberrant MZ that may affect responses to blood-borne pathogens and peripheral B-cell tolerance.
造血细胞的发育依赖于动态的肌动蛋白细胞骨架。在此,我们证明,在威斯科特-奥尔德里奇综合征中发生突变的细胞骨架调节因子WASP的表达,为天然调节性T细胞、自然杀伤性T细胞、CD4(+)和CD8(+) T淋巴细胞、边缘区(MZ)B细胞、MZ巨噬细胞和血小板的发育提供了选择性优势。为了确定MZ B细胞和MZ巨噬细胞对MZ发育的相对贡献,我们构建了野生型和WASP缺陷型骨髓嵌合小鼠,其MZ完全恢复。然而,即使存在MZ巨噬细胞,只有10%的MZ B细胞来源于WASP缺陷型。我们发现,WASP缺陷型MZ B细胞在体内过度增殖,并且对1-磷酸鞘氨醇(一种对MZ B细胞定位至关重要的趋化因子)无反应。WASP(-/-)小鼠MZ区室的异常导致金黄色葡萄球菌摄取异常,并导致对TNP-菲可的免疫反应降低。此外,WASP缺陷型小鼠的“天然”IgM抗体水平升高。我们的研究结果表明,WASP调节造血细胞的发育和功能。我们证明,WASP缺陷会导致异常的MZ,这可能会影响对血源性病原体的反应和外周B细胞的耐受性。