与 XMEN 相关的儿童系统性 EBV 阳性 T 细胞淋巴瘤:两例病例报告和文献综述。

IF 0.9 4区 医学 Q4 HEMATOLOGY
Ping Cao, Xiao Zhang, Yang Fu, Hongsheng Wang, Yi Yu, Xiaohua Zhu, Junye Jiang, Xiaowen Zhai
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引用次数: 0

摘要

伴有镁缺陷、爱泼斯坦-巴氏病毒(EBV)感染和肿瘤的X连锁免疫缺陷症(XMEN)是一种极其罕见的先天性免疫错误(IEI),由X连锁隐性遗传和MAGT1基因的功能缺失突变引起,导致镁离子通道缺陷。本文报告了2例与XMEN相关的儿童系统性EB病毒阳性T细胞淋巴瘤(SETLC),这些病例以前从未报道过。他们家族的全外显子组测序(WES)发现了之前未报道过的MAGT1基因突变(c.77T>C,p.I26T;c.956-957del:p.Ser319Tyrfs),这些突变遗传自他们的母亲。这些突变扩大了 XMEN 病的基因突变范围。我们强调了 MAGT1 突变基因检测在 SETLC 初步诊断中的重要性。我们还回顾了有关这种不常见 IEI 的文献。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
XMEN-associated Systemic EBV-positive T-cell Lymphoma of Childhood: Report of Two Cases and Literature Review.

X-linked immunodeficiency with magnesium defect, Epstein-Barr virus (EBV) infection, and neoplasia (XMEN) is an extremely rare inborn error of immunity (IEI) caused by X-linked recessive inheritance and loss-of-function mutations in the MAGT1 gene, resulting in magnesium ion channel defects. This article reports 2 cases of systemic EBV-positive T-cell Lymphoma of childhood (SETLC) associated with XMEN, which have not been reported before. Whole exome sequencing (WES) in their family revealed previously unreported MAGT1 gene mutations (c.77T>C, p.I26T; c.956-957del: p.Ser319Tyrfs) inherited from their mothers. These mutations expand the spectrum of gene mutations in XMEN disease. The importance of genetic testing for MAGT1 mutations in the initial diagnosis of SETLC was emphasized. We also review the literature on this uncommon IEI.

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来源期刊
CiteScore
1.90
自引率
8.30%
发文量
415
审稿时长
2.5 months
期刊介绍: ​Journal of Pediatric Hematology/Oncology (JPHO) reports on major advances in the diagnosis and treatment of cancer and blood diseases in children. The journal publishes original research, commentaries, historical insights, and clinical and laboratory observations.
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