Toyama Daisuke, Matsuno Ryosuke, Sugishita Yumiko, Kaneko Ryota, Okamoto Naoko, Koganesawa Masaya, Fujita Sachio, Akiyama Kosuke, Isoyama Keiichi, Yamamoto Shohei
Department of Pediatrics, Showa University Fujigaoka Hospital, Yokohama, Japan.
Case Rep Hematol. 2020 Feb 12;2020:1378056. doi: 10.1155/2020/1378056. eCollection 2020.
Prognosis in pediatric patients with refractory/relapsed acute myeloid leukemia (AML) is grim, and there is no standard treatment for such patients. Combined treatment with intensive chemotherapy and gemtuzumab ozogamicin (GO), a monoclonal anti-CD33 antibody conjugated with calicheamicin, is useful as reinduction therapy in refractory/relapsed AML. Here, we describe three cases of pediatric refractory/relapsed AML that were successfully managed with FLAG-IDA (fludarabine, cytarabine, granulocyte colony-stimulating factor, and idarubicin), with or without GO, as reinduction therapy before a KIR-ligand-mismatched cord blood transplant. This strategy relies on the fact that killer cell immunoglobulin-like receptors (KIR) on cord blood natural killer (NK) cells recognize human leukocyte antigen (HLA) class I alleles, and that donor KIR-ligand incompatibility may be associated with lower incidence of relapse and improved survival in AML, as cells that lack these inhibitory HLA ligands can activate NK cells. All three patients are currently alive and have been disease-free for 24-65 months, although one patient developed severe sinusoidal obstructive syndrome (SOS). Thus, our strategy can result in excellent outcomes in pediatric patients with refractory/relapsed AML.
难治性/复发性急性髓系白血病(AML)患儿的预后严峻,且此类患者尚无标准治疗方法。强化化疗与吉妥珠单抗奥唑米星(GO,一种与卡奇霉素偶联的抗CD33单克隆抗体)联合治疗,可用作难治性/复发性AML的再诱导治疗。在此,我们描述了3例小儿难治性/复发性AML病例,在进行KIR配体不匹配的脐血移植前,采用FLAG-IDA(氟达拉滨、阿糖胞苷、粒细胞集落刺激因子和伊达比星)进行再诱导治疗,其中1例联合使用了GO。该策略基于以下事实:脐血自然杀伤(NK)细胞上的杀伤细胞免疫球蛋白样受体(KIR)可识别人类白细胞抗原(HLA)I类等位基因,并且供体KIR配体不相容可能与AML复发率降低和生存率提高相关,因为缺乏这些抑制性HLA配体的细胞可激活NK细胞。所有3例患者目前均存活,且已无病生存24 - 65个月,不过有1例患者发生了严重的肝窦阻塞综合征(SOS)。因此,我们的策略可使小儿难治性/复发性AML患者获得良好预后。