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急性全身性巨细胞病毒感染触发噬血细胞性淋巴组织细胞增生症年轻女性 GATA2 缺陷症的诊断。

Diagnosis of GATA2 Deficiency in a Young Woman with Hemophagocytic Lymphohistiocytosis Triggered by Acute Systemic Cytomegalovirus Infection.

机构信息

Department of Internal Medicine, Morristown Medical Center, Morristown, NJ, USA.

Department of Hematology/Oncology, Morristown Medical Center, Morristown, NJ, USA.

出版信息

Am J Case Rep. 2021 Mar 8;22:e927087. doi: 10.12659/AJCR.927087.

DOI:10.12659/AJCR.927087
PMID:33684095
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7959100/
Abstract

BACKGROUND Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening disease characterized by an intense immunologic response that results in multiorgan dysfunction. It typically manifests as a result of a familial genetic immunodeficiency disorder or secondary to a trigger such as an infection, malignancy, or autoimmune disease. The major factors involved in the development of the disease are an individual's genetic propensity to develop HLH, such as rare associated mutations, or inflammatory processes that trigger the immune system to go haywire. CASE REPORT Before the COVID-19 pandemic, a 22-year-old woman with a history of congenital absence of the right kidney, right-sided hearing loss, and leukopenia presented with a 3-week history of generalized malaise, fever, chest pain, cough, and shortness of breath. She developed an acute systemic cytomegalovirus infection further complicated by HLH. Based on her history and clinical course, an underlying primary immunodeficiency was suspected. An immunodeficiency gene panel revealed a monoallelic mutation in GATA2, a gene that encodes zinc-transcription factors responsible for the regulation of hematopoiesis. CONCLUSIONS GATA2 deficiency encompasses a large variety of mutations in the GATA2 gene and leads to disorders associated with hematologic and immunologic manifestations of monocytopenia and B-, and natural killer-cell deficiency. Over time, affected individuals are at high risk of developing life-threatening infections and serious hematologic complications, such as myelodysplastic syndromes and/or leukemias. We aimed to illustrate the importance of identifying an underlying genetic disorder associated with secondary HLH to help guide acute and long-term management.

摘要

背景

噬血细胞性淋巴组织细胞增生症(HLH)是一种危及生命的疾病,其特征是强烈的免疫反应导致多器官功能障碍。它通常是家族遗传性免疫缺陷疾病的结果,或者继发于感染、恶性肿瘤或自身免疫性疾病等诱因。疾病发展的主要因素是个体发生 HLH 的遗传倾向,如罕见的相关突变,或触发免疫系统失控的炎症过程。

病例报告

在 COVID-19 大流行之前,一名 22 岁女性,有先天性右肾缺失、右侧听力丧失和白细胞减少症病史,出现了 3 周的全身不适、发热、胸痛、咳嗽和呼吸急促。她发生了急性全身性巨细胞病毒感染,进一步并发噬血细胞性淋巴组织细胞增生症。根据她的病史和临床过程,怀疑存在潜在的原发性免疫缺陷。免疫缺陷基因谱显示 GATA2 基因的单等位基因突变,该基因编码锌转录因子,负责造血的调节。

结论

GATA2 缺陷包括 GATA2 基因的多种突变,导致与单核细胞减少症和 B 细胞、自然杀伤细胞缺陷相关的血液学和免疫学表现的疾病。随着时间的推移,受影响的个体有发生危及生命的感染和严重血液学并发症的高风险,如骨髓增生异常综合征和/或白血病。我们旨在阐明识别与继发性 HLH 相关的潜在遗传疾病的重要性,以帮助指导急性和长期管理。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d1e/7959100/909fddd517b5/amjcaserep-22-e927087-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d1e/7959100/38aa4e8fafbe/amjcaserep-22-e927087-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d1e/7959100/909fddd517b5/amjcaserep-22-e927087-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d1e/7959100/38aa4e8fafbe/amjcaserep-22-e927087-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d1e/7959100/909fddd517b5/amjcaserep-22-e927087-g002.jpg

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