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两名新型 DOCK2 基因突变患者存在多种免疫缺陷,导致反复发生多种感染,包括活减毒病毒疫苗感染。

Multiple Immune Defects in Two Patients with Novel DOCK2 Mutations Result in Recurrent Multiple Infection Including Live Attenuated Virus Vaccine.

机构信息

National Clinical Research Center for Child Health and Disorders (Chongqing), Children's Hospital of Chongqing Medical University, Chongqing, China.

Ministry of Education Key Laboratory of Child Development and Disorders, Children's Hospital of Chongqing Medical University, Chongqing, China.

出版信息

J Clin Immunol. 2023 Aug;43(6):1193-1207. doi: 10.1007/s10875-023-01466-y. Epub 2023 Mar 22.

Abstract

The dedicator of cytokinesis 2(DOCK2) protein, an atypical guanine nucleotide exchange factor (GEFs), is a member of the DOCKA protein subfamily. DOCK2 protein deficiency is characterized by early-onset lymphopenia, recurrent infections, and lymphocyte dysfunction, which was classified as combined immune deficiency with neutrophil abnormalities as well. The only cure is hematopoietic stem cell transplantation. Here, we report two patients harboring four novel DOCK2 mutations associated with recurrent infections including live attenuated vaccine-related infections. The patient's condition was partially alleviated by symptomatic treatment or intravenous immunoglobulin. We also confirmed defects in thymic T cell output and T cell proliferation, as well as aberrant skewing of T/B cell subset TCR-Vβ repertoires. In addition, we noted neutrophil defects, the weakening of actin polymerization, and BCR internalization under TCR/BCR activation. Finally, we found that the DOCK2 protein affected antibody affinity although with normal total serum immunoglobulin. The results reported herein expand the clinical phenotype, the pathogenic DOCK2 mutation database, and the immune characteristics of DOCK2-deficient patients.

摘要

细胞分裂蛋白 2(DOCK2)的配体,一种非典型的鸟嘌呤核苷酸交换因子(GEFs),是 DOCKA 蛋白亚家族的成员。DOCK2 蛋白缺乏的特征是早期发生的淋巴细胞减少、反复感染和淋巴细胞功能障碍,这也被归类为伴有中性粒细胞异常的联合免疫缺陷。唯一的治疗方法是造血干细胞移植。在这里,我们报告了两名患者,他们携带了四个与反复感染相关的新型 DOCK2 突变,包括活减毒疫苗相关感染。患者的病情通过对症治疗或静脉注射免疫球蛋白得到部分缓解。我们还证实了胸腺 T 细胞输出和 T 细胞增殖缺陷,以及 T/B 细胞亚群 TCR-Vβ 库的异常偏向。此外,我们注意到中性粒细胞缺陷、TCR/BCR 激活下肌动蛋白聚合减弱和 BCR 内化。最后,我们发现 DOCK2 蛋白尽管总血清免疫球蛋白正常,但影响了抗体亲和力。本报告的结果扩展了 DOCK2 缺陷患者的临床表型、致病性 DOCK2 突变数据库和免疫特征。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d55c/10032263/f4cdfd41843b/10875_2023_1466_Fig1_HTML.jpg

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