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匈牙利的家族性急性髓系白血病和骨髓增生异常综合征

Familial Acute Myeloid Leukemia and Myelodysplasia in Hungary.

作者信息

Király Attila Péter, Kállay Krisztián, Gángó Ambrus, Kellner Ádám, Egyed Miklós, Szőke Anita, Kiss Richárd, Vályi-Nagy István, Csomor Judit, Matolcsy András, Bödör Csaba

机构信息

MTA-SE Lendület Molecular Oncohematology Research Group, 1st Department of Pathology and Experimental Cancer Research, Semmelweis University, Budapest, Hungary.

Pediatric Hematology and Stem Cell Transplantation Unit, United St. István and St. László Hospital, Budapest, Hungary.

出版信息

Pathol Oncol Res. 2018 Jan;24(1):83-88. doi: 10.1007/s12253-017-0216-4. Epub 2017 Mar 29.

Abstract

Although genetic predisposition to haematological malignancies has long been known, genetic testing is not yet the part of the routine diagnostics. In the last ten years, next generation sequencing based studies identified novel germline mutations in the background of familial aggregation of certain haematologic disorders including myelodysplastic syndromes (MDS) and acute myeloid leukaemia (AML). This is supported by the fact that the myeloid neoplasms with genetic predisposition represent a new category in the revised 2016 World Health Organization classification. According to the new classification, these disorders are subdivided based on the clinical and genetic features, including myeloid neoplasms with germline predisposition alone, or with pre-existing platelet disorder, cytopaenias or other organ failures. The predisposing genetic factors include mutations in the RUNX1, CEBPA, GATA2, ANKRD26, ETV6, DDX41, TERC or TERT and SRP72 genes. The genes affected in these syndromes are important regulators of haemopoiesis and are frequently implicated in leukaemogenesis, providing deeper insight into the understanding of normal and malignant haemopoiesis. Despite the growing knowledge of germline predisposing events in the background of familial myeloid malignancies, the germline genetic component is still unknown in a subset of these pedigrees. Here, we present the first study of inherited myeloid malignancies in Hungary. We identified three families with apparent clustering of myeloid malignancies with nine affected individuals across these pedigrees. All tested individuals were negative for CEBPA, GATA2, RUNX1, ANKRD26, ETV6, DDX41, TERC or TERT and SRP72 mutations, suggesting the presence of so far unidentified predisposing mutations.

摘要

尽管血液系统恶性肿瘤的遗传易感性早已为人所知,但基因检测尚未成为常规诊断的一部分。在过去十年中,基于下一代测序的研究在某些血液系统疾病(包括骨髓增生异常综合征(MDS)和急性髓系白血病(AML))的家族聚集背景下发现了新的种系突变。这得到了以下事实的支持:具有遗传易感性的髓系肿瘤在2016年世界卫生组织修订分类中代表一个新类别。根据新分类,这些疾病根据临床和遗传特征进行细分,包括仅具有种系易感性的髓系肿瘤,或伴有既往血小板疾病、血细胞减少或其他器官功能衰竭的髓系肿瘤。易感遗传因素包括RUNX1、CEBPA、GATA2、ANKRD26、ETV6、DDX41、TERC或TERT以及SRP72基因中的突变。这些综合征中受影响的基因是造血的重要调节因子,经常与白血病发生有关,为深入理解正常和恶性造血提供了线索。尽管在家族性髓系恶性肿瘤背景下对种系易感事件的认识不断增加,但在这些家系的一部分中,种系遗传成分仍然未知。在此,我们展示了匈牙利遗传性髓系恶性肿瘤的第一项研究。我们确定了三个家族,这些家族中髓系恶性肿瘤明显聚集,这些家系中有九名受影响个体。所有检测个体的CEBPA、GATA2、RUNX1、ANKRD26、ETV6、DDX41、TERC或TERT以及SRP72突变均为阴性,这表明存在迄今尚未鉴定的易感突变。

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