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异基因造血干细胞移植后供体细胞源性急性早幼粒细胞白血病。

Donor cell-derived acute promyelocytic leukemia after allogeneic hematopoietic stem cell transplantation.

机构信息

Laboratoire d'Hématologie, CHU d'Angers, Angers, France.

Fédération Hospitalo-Universitaire 'Grand Ouest Against Leukemia' (FHU GOAL), Angers, France.

出版信息

Eur J Haematol. 2018 Oct;101(4):570-574. doi: 10.1111/ejh.13143. Epub 2018 Sep 3.

Abstract

Donor cell leukemia (DCL) is an infrequent complication after allogeneic hematopoietic stem cell transplantation (HSCT). Its true incidence is difficult to assess, although improvements in chimerism studies contributed to a better diagnosis of DCL. We report two rare cases of donor cell-derived acute promyelocytic leukemia (APL). To our knowledge, only two cases have been described in the literature. Here, we report one male and one female patients with acute myeloid leukemia (AML), who developed an APL in donor cells after HSCT. The latency between HSCT and DCL was 279 and 43 months, respectively. Fluorescent in situ hybridation and chimerism monitoring analysis proved the donor origin of APL. Surprisingly, donor lymphocyte infusion provided a hematological response during 19 months in the female patient. The mechanisms associated with pathogenesis of DCL are unclear and seem to be multifactorial. Increasing worldwide allogeneic hematopoietic stem cell transplantation activity and potentially the age of donor could explain the increasing incidence of DCL in the future. It is highlighted that long-term follow up of recipients will allow to report all cases of DCL, to clarify the genetic landscape and factors which contribute to DCL, to understand the response to DLI.

摘要

供体细胞白血病(DCL)是异基因造血干细胞移植(HSCT)后一种罕见的并发症。虽然嵌合研究的改进有助于更好地诊断 DCL,但要准确评估其真实发病率仍很困难。我们报告了两例罕见的供体细胞衍生的急性早幼粒细胞白血病(APL)病例。据我们所知,文献中仅描述过两例。在此,我们报告了两例男性和女性的急性髓系白血病(AML)患者,他们在 HSCT 后供体细胞中发生了 APL。HSCT 与 DCL 之间的潜伏期分别为 279 个月和 43 个月。荧光原位杂交和嵌合监测分析证明了 APL 的供体来源。令人惊讶的是,在女性患者中,供者淋巴细胞输注在 19 个月内提供了血液学反应。与 DCL 发病机制相关的机制尚不清楚,似乎是多因素的。全球异基因造血干细胞移植活动的增加以及供者年龄的增加可能解释了未来 DCL 发病率的增加。需要强调的是,对受者进行长期随访将能够报告所有 DCL 病例,阐明 DCL 的遗传特征和相关因素,以及了解对 DLI 的反应。

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