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唐氏综合征的短暂性白血病(短暂性骨髓增殖性疾病,短暂性异常髓系造血)

Transient leukemia (transient myeloproliferative disorder, transient abnormal myelopoiesis) of Down syndrome.

作者信息

Brink David S

机构信息

Department of Pathology, Saint Louis University School of Medicine, Saint Louis, MO 63104-1003, USA.

出版信息

Adv Anat Pathol. 2006 Sep;13(5):256-62. doi: 10.1097/01.pap.0000213039.93328.44.

Abstract

Transient leukemia of Down syndrome (DS-TL), also known as transient myeloproliferative disorder of Down syndrome (DS) and transient abnormal myelopoiesis of DS, occurs in approximately 10% of DS neonates and in phenotypically normal neonates with trisomy 21 mosaicism. In DS-TL, peripheral blood analysis shows variable numbers of blasts and, usually, thrombocytopenia; other cytopenias are uncommon. Bone marrow characteristics of DS-TL are, likewise, variable, though (in contrast to other leukemias) the bone marrow blast differential can be lower than the peripheral blood blast differential. The blasts of DS-TL typically show light microscopic, ultrastructural, and flow cytometric evidence of megakaryocyte differentiation. DS-TL neonates have a approximately 15% risk of developing potentially fatal liver disease and show <10% incidence of hydrops fetalis. Additional manifestations of DS-TL include cutaneous involvement, hyperviscosity, myelofibrosis, cardiopulmonary failure, splenomegaly, and spleen necrosis. Despite its typical transient nature, 20% to 30% of DS-TL patients develop overt (nontransient) acute leukemia, usually within 3 years and typically of the M7 phenotype (acute megakaryoblastic leukemia). The pathogenesis of DS-TL (and of subsequent acute leukemia) involves mutation of GATA1 (on chromosome X), which normally encodes a transcription factor integral to normal development of erythroid, megakaryocytic, and basophilic/mast cell lines. The pathogenetic role of trisomy 21 in DS-TL is unclear. Though indications for chemotherapy in DS-TL have not been firmly established, the blasts of DS-TL are sensitive to low-dose cytosine arabinoside.

摘要

唐氏综合征相关短暂性白血病(DS-TL),也被称为唐氏综合征相关短暂性骨髓增殖性疾病(DS-TMD)以及唐氏综合征相关短暂性异常髓系造血,约10%的唐氏综合征新生儿以及表型正常的21号染色体三体嵌合体新生儿会发病。在DS-TL中,外周血分析显示有数量不等的原始细胞,通常还有血小板减少;其他血细胞减少并不常见。DS-TL的骨髓特征同样各异,不过(与其他白血病不同)骨髓原始细胞比例可能低于外周血原始细胞比例。DS-TL的原始细胞通常在光镜、超微结构以及流式细胞术检查中显示有巨核细胞分化的证据。DS-TL新生儿有大约15%的风险会发展为潜在致命性肝病,且胎儿水肿的发生率低于10%。DS-TL的其他表现包括皮肤受累、血液黏滞度增高、骨髓纤维化、心肺衰竭、脾肿大以及脾坏死。尽管其通常具有短暂性,但20%至30%的DS-TL患者会发展为明显的(非短暂性)急性白血病,通常在3年内发病,且典型的为M7表型(急性巨核细胞白血病)。DS-TL(以及后续急性白血病)的发病机制涉及GATA1(位于X染色体上)突变,该基因通常编码一种对红系、巨核系以及嗜碱性/肥大细胞系正常发育至关重要的转录因子。21号染色体三体在DS-TL中的致病作用尚不清楚。尽管DS-TL化疗的指征尚未明确确立,但DS-TL的原始细胞对低剂量阿糖胞苷敏感。

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