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