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患有血液学表现的种系GATA2突变患者中与骨髓增生异常综合征相关的突变。

MDS-associated mutations in germline GATA2 mutated patients with hematologic manifestations.

作者信息

McReynolds Lisa J, Yang Yanqin, Yuen Wong Hong, Tang Jingrong, Zhang Yubo, Mulé Matthew P, Daub Janine, Palmer Cindy, Foruraghi Ladan, Liu Qingguo, Zhu Jun, Wang Weixin, West Robert R, Yohe Marielle E, Hsu Amy P, Hickstein Dennis D, Townsley Danielle M, Holland Steven M, Calvo Katherine R, Hourigan Christopher S

机构信息

Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.

DNA Sequencing and Genomics Core, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD, USA.

出版信息

Leuk Res. 2019 Jan;76:70-75. doi: 10.1016/j.leukres.2018.11.013. Epub 2018 Dec 4.

Abstract

Germline mutation in GATA2 can lead to GATA2 deficiency characterized by a complex multi-system disorder that can present with many manifestations including variable cytopenias, bone marrow failure, myelodysplastic syndrome/acute myeloid leukemia (MDS/AML), and severe immunodeficiency. Penetrance and expressivity within families is often variable. There is a spectrum of bone marrow disease in symptomatic cytopenic patients ranging from hypocellular marrows without overt dysplasia to those with definitive MDS, AML, or chronic myelomonocytic leukemia. Relatives of probands with the same mutations may demonstrate minimal disease manifestations and normal marrows. A comprehensive clinical, hematological and genetic assessment of 25 patients with germline GATA2 mutation was performed. MDS-associated mutations were identified in symptomatic GATA2 patients both with overt MDS and in those with hypocellular/aplastic bone marrows without definitive dysplasia. Healthy relatives of probands harboring the same germline GATA2 mutations had essentially normal marrows that were overall devoid of MDS-associated mutations. The findings suggest that abnormal clonal hematopoiesis is a common event in symptomatic germline mutated GATA2 patients with MDS and also in those with hypocellular marrows without overt morphologic evidence of dysplasia, possibly indicating a pre-MDS stage warranting close monitoring for disease progression.

摘要

GATA2基因的种系突变可导致GATA2缺乏症,其特征为一种复杂的多系统疾病,可表现出多种症状,包括不同程度的血细胞减少、骨髓衰竭、骨髓增生异常综合征/急性髓系白血病(MDS/AML)以及严重免疫缺陷。家族中的外显率和表现度通常存在差异。有症状的血细胞减少患者存在一系列骨髓疾病,从无明显发育异常的细胞减少性骨髓到明确诊断为MDS、AML或慢性粒单核细胞白血病的骨髓。携带相同突变的先证者亲属可能仅有轻微的疾病表现且骨髓正常。对25例GATA2种系突变患者进行了全面的临床、血液学和遗传学评估。在有明显MDS的有症状GATA2患者以及无明确发育异常的细胞减少/再生障碍性骨髓患者中均发现了与MDS相关的突变。携带相同GATA2种系突变的先证者的健康亲属骨髓基本正常,总体上没有与MDS相关的突变。这些发现表明,异常克隆性造血在有症状的GATA2种系突变且患有MDS的患者以及无明显形态学发育异常证据的细胞减少性骨髓患者中是常见事件,这可能表明处于MDS前期阶段,需要密切监测疾病进展。

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