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1型CD36缺乏症患者成功进行异基因造血干细胞移植:病例研究及文献综述

Successful allogeneic hematopoietic stem cell transplantation in a patient with type I CD36 deficiency: a case study and literature review.

作者信息

Okuyama Shuhei, Sumi Masahiko, Ishikawa Ryuto, Shishido Tsutomu, Koyama Daisuke, Ueki Toshimitsu, Takahashi Daisuke, Kobayashi Hironori, Kobayashi Hikaru, Tsuno Nelson Hirokazu

机构信息

Department of Hematology, Nagano Red Cross Hospital, Nagano, Japan.

Department of Hematology, Yamagata Prefectural Central Hospital, 1800 Aoyagi, Yamagata-Shi, Yamagata, 990-2292, Japan.

出版信息

Int J Hematol. 2023 Nov;118(5):656-660. doi: 10.1007/s12185-023-03637-4. Epub 2023 Jul 25.

Abstract

BACKGROUND

CD36-deficient individuals may produce anti-CD36 antibodies through antigenic exposure to CD36, in situations including blood transfusions. Therefore, allogeneic hematopoietic stem cell transplantation (HSCT) from CD36-positive donors to CD36-negative patients remains a challenge.

CASE REPORT

A 64-year-old man with acute myeloid leukemia became refractory to platelet transfusions during chemotherapy. Anti-CD36 antibodies without anti-HLA antibodies were detected in serum, and the absence of CD36 expression on platelets and monocytes confirmed type I CD36 deficiency. The patient achieved complete remission, and received maintenance therapy with CD36-negative platelet transfusions. However, he relapsed soon afterward, and thus underwent peripheral blood stem cell transplantation (PBSCT) from a CD36-positive unrelated donor. The anti-CD36 antibody titer had decreased before the transplant, and the PBSCT-course was uneventful. The patient has been well without any complications associated with CD36 status mismatch.

DISCUSSION

The few reports of allogeneic HSCT in patients with CD36 deficiency have suggested that anti-CD36 antibodies could be involved in several post-transplant complications, such as delayed platelet recovery, transfusion refractoriness, and transfusion-related acute lung injury. Our present case confirmed that stem cell transplantation from CD36-positive donors to negative patients is feasible, when it includes careful prior assessment of anti-CD36 antibody titers and interventions to attenuate them.

摘要

背景

CD36缺陷个体可能通过输血等情况下对CD36的抗原暴露产生抗CD36抗体。因此,从CD36阳性供体向CD36阴性患者进行异基因造血干细胞移植(HSCT)仍然是一项挑战。

病例报告

一名64岁急性髓系白血病男性患者在化疗期间对血小板输注产生耐受。血清中检测到无抗HLA抗体的抗CD36抗体,血小板和单核细胞上CD36表达缺失证实为I型CD36缺陷。患者实现完全缓解,接受CD36阴性血小板输注维持治疗。然而,他随后很快复发,因此接受了来自CD36阳性非亲属供体的外周血干细胞移植(PBSCT)。移植前抗CD36抗体滴度已降低,PBSCT过程顺利。患者情况良好,未出现与CD36状态不匹配相关的任何并发症。

讨论

关于CD36缺陷患者异基因HSCT的少数报告表明,抗CD36抗体可能与几种移植后并发症有关,如血小板恢复延迟、输血耐受和输血相关急性肺损伤。我们目前的病例证实,从CD36阳性供体向阴性患者进行干细胞移植是可行的,前提是包括对抗CD36抗体滴度进行仔细的预先评估并采取措施降低其水平。

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