Department of Rheumatology and Immunology, Children's Hospital of Chongqing Medical University, National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Child Infection and Immunity, Chongqing, China.
BMC Pediatr. 2023 Apr 26;23(1):195. doi: 10.1186/s12887-023-03975-6.
Zeta(ζ)-Chain Associated Protein Kinase 70 kDa (ZAP-70) deficiency is a rare autosomal recessive primary immunodeficiency disease. Little is known about this disease. In this study, we report two patients to extend the range of clinical phenotypes and immunophenotypes associated with ZAP-70 mutations. We describe the clinical, genetic, and immunological phenotypes of two patients with ZAP-70 deficiency in China, and the data are also compared with the literature. Case 1 presented with leaky severe combined immunodeficiency with low to the absence of CD8 + T cells, while case 2 suffered from a recurrent respiratory infection and had a past medical history of non-EBV-associated Hodgkin's lymphoma. Sequencing revealed novel compound heterozygous mutations in ZAP-70 of these patients. Case 2 is the second ZAP-70 patient presenting a normal CD8 + T cell number. These two cases have been treated with hematopoietic stem cell transplantation. Selective CD8 + T cell loss is an essential feature of the immunophenotype of ZAP-70 deficiency patients, but there are exceptions. Hematopoietic stem cell transplantation can provide excellent long-term immune function and resolution of clinical problems.
Zeta(ζ)-链关联蛋白激酶 70kDa(ZAP-70)缺陷是一种罕见的常染色体隐性原发性免疫缺陷病。目前对此病的了解甚少。在本研究中,我们报告了两例患者,以扩展与 ZAP-70 突变相关的临床表型和免疫表型范围。我们描述了中国两名 ZAP-70 缺陷患者的临床、遗传和免疫学表型,并将数据与文献进行了比较。病例 1 表现为渗漏性严重联合免疫缺陷,CD8+T 细胞数量低至缺失,而病例 2 则患有复发性呼吸道感染,并曾患有非 EBV 相关霍奇金淋巴瘤。测序揭示了这些患者 ZAP-70 的新型复合杂合突变。病例 2 是第二个表现为正常 CD8+T 细胞数量的 ZAP-70 患者。这两例患者均接受了造血干细胞移植治疗。选择性 CD8+T 细胞缺失是 ZAP-70 缺陷患者免疫表型的一个重要特征,但也有例外。造血干细胞移植可以提供出色的长期免疫功能和解决临床问题。