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骨髓衰竭综合征的细胞遗传学管理:来自法语血液细胞遗传学组 (GFCH) 的指南。

Cytogenetics in the management of bone marrow failure syndromes: Guidelines from the Groupe Francophone de Cytogénétique Hématologique (GFCH).

机构信息

Laboratoire d'Hématologie, Unité de Cytogénétique, Hôpital Saint-Louis, Assistance Publique Hôpitaux de Paris (APHP), 75475, Paris Cedex 10, France.

Oncobiologie Génétique Bioinformatique UF Cytogénétique et Génétique Moléculaire, CHU de Besançon, Hôpital Minjoz, 25030, Besançon, France.

出版信息

Curr Res Transl Med. 2023 Oct-Dec;71(4):103423. doi: 10.1016/j.retram.2023.103423. Epub 2023 Oct 18.

Abstract

Bone marrow failure syndromes are rare disorders characterized by bone marrow hypocellularity and resultant peripheral cytopenias. The most frequent form is acquired, so-called aplastic anemia or idiopathic aplastic anemia, an auto-immune disorder frequently associated with paroxysmal nocturnal hemoglobinuria, whereas inherited bone marrow failure syndromes are related to pathogenic germline variants. Among newly identified germline variants, GATA2 deficiency and SAMD9/9L syndromes have a special significance. Other germline variants impacting biological processes, such as DNA repair, telomere biology, and ribosome biogenesis, may cause major syndromes including Fanconi anemia, dyskeratosis congenita, Diamond-Blackfan anemia, and Shwachman-Diamond syndrome. Bone marrow failure syndromes are at risk of secondary progression towards myeloid neoplasms in the form of myelodysplastic neoplasms or acute myeloid leukemia. Acquired clonal cytogenetic abnormalities may be present before or at the onset of progression; some have prognostic value and/or represent somatic rescue mechanisms in inherited syndromes. On the other hand, the differential diagnosis between aplastic anemia and hypoplastic myelodysplastic neoplasm remains challenging. Here we discuss the value of cytogenetic abnormalities in bone marrow failure syndromes and propose recommendations for cytogenetic diagnosis and follow-up.

摘要

骨髓衰竭综合征是一种罕见疾病,其特征为骨髓细胞减少,导致外周血细胞减少。最常见的形式是获得性的,即所谓的再生障碍性贫血或特发性再生障碍性贫血,这是一种自身免疫性疾病,常与阵发性睡眠性血红蛋白尿症相关,而遗传性骨髓衰竭综合征与致病性种系变异有关。在新鉴定的种系变异中,GATA2 缺乏症和 SAMD9/9L 综合征具有特殊意义。其他影响生物学过程的种系变异,如 DNA 修复、端粒生物学和核糖体生物发生,可能导致包括范可尼贫血、先天性角化不良、 Diamond-Blackfan 贫血和 Shwachman-Diamond 综合征在内的主要综合征。骨髓衰竭综合征有向骨髓增生异常或急性髓系白血病等髓系肿瘤继发进展的风险。获得性克隆细胞遗传学异常可能在进展前或进展时出现;一些具有预后价值,或代表遗传性综合征中的体细胞拯救机制。另一方面,再生障碍性贫血和低增生性骨髓增生异常之间的鉴别诊断仍然具有挑战性。在此,我们讨论了细胞遗传学异常在骨髓衰竭综合征中的价值,并提出了细胞遗传学诊断和随访的建议。

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