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患者患有 B 淋巴母细胞白血病和 EBV 相关弥漫性大 B 细胞淋巴瘤,存在 DOCK8 复合杂合突变。

Compound Heterozygous DOCK8 Mutations in a Patient with B Lymphoblastic Leukemia and EBV-Associated Diffuse Large B Cell Lymphoma.

机构信息

Department of Hematology, Children's Hospital of Orange County, 1201 W. La Veta Avenue, Orange, CA, 92868, USA.

Department of Pediatrics, University of California at Irvine, Orange, CA, USA.

出版信息

J Clin Immunol. 2019 Aug;39(6):592-595. doi: 10.1007/s10875-019-00663-y. Epub 2019 Jul 2.

Abstract

Mutations in Dedicator of cytokinesis 8 (DOCK8) are a rare cause of combined immunodeficiency associated with atopy, infectious susceptibility, and risk for malignancy. We describe a 22-year-old male with a diagnosis of B cell lymphoblastic leukemia followed by Epstein-Barr virus (EBV)-associated diffuse large B cell lymphoma (DLBCL) with compound heterozygous mutations in DOCK8 and normal intracellular DOCK8 protein expression. Here, B cell lymphoblastic leukemia followed by EBV-associated DLBCL led to the discovery of DOCK8 deficiency. For instances of high clinical suspicion despite normal DOCK8 protein expression, additional functional testing is critical to make a diagnosis. Understanding the spectrum of DOCK8 mutants and their phenotypes will improve our understanding of DOCK8 deficiency.

摘要

细胞分裂定向因子 8(DOCK8)突变是一种罕见的联合免疫缺陷病的病因,常伴有特应性、易感性感染和恶性肿瘤风险。我们描述了一名 22 岁男性,患有 B 细胞淋巴母细胞白血病,随后发生 EBV 相关弥漫性大 B 细胞淋巴瘤(DLBCL),存在 DOCK8 的复合杂合突变和正常的细胞内 DOCK8 蛋白表达。在这里,B 细胞淋巴母细胞白血病继发 EBV 相关 DLBCL 导致了 DOCK8 缺陷的发现。对于尽管 DOCK8 蛋白表达正常但仍有高度临床怀疑的情况,额外的功能测试对于做出诊断至关重要。了解 DOCK8 突变体及其表型的范围将有助于我们更好地理解 DOCK8 缺陷。

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