Mori Jinichi, Oshima Kumi, Kimura Satoshi, Ikezoe Takayuki
Department of Hematology, Jyoban Hospital, Tokiwa Foundation, Fukushima, Japan.
Patient Safety Division, QI Center, St. Luke's International Hospital, Tokyo, Japan.
Case Rep Hematol. 2020 Jul 27;2020:8822172. doi: 10.1155/2020/8822172. eCollection 2020.
T-cell prolymphocytic leukemia (T-PLL) is a rare hematologic cancer with a dismal prognosis. Although a small number of patients have central nervous system (CNS) involvement, a standard treatment approach for these patients has not been established. Herein, we present a case of T-PLL with CNS involvement that was treated with a higher dose of intrathecal alemtuzumab than that previously reported. A 66-year-old man who had T-PLL with CNS involvement was admitted to our hospital. Intravenously administered alemtuzumab, a monoclonal antibody against the CD52 antigen, successfully reduced leukemia cells in peripheral blood; however, intrathecal treatment with methotrexate, cytarabine, and prednisone had a limited effect on the CNS involvement. Therefore, we intrathecally injected alemtuzumab as an experimental treatment. Although we escalated the dose of intrathecal alemtuzumab up to 5 mg, no adverse reaction was noted; however, this treatment did not completely clear the leukemia cells from the patient's cerebrospinal fluid (CSF). We performed whole brain and whole spinal irradiation therapies and subsequently performed allogeneic transplantation from a human leukocyte antigen-matched sibling donor with a conditioning regimen containing total body irradiation. At 21 days after transplantation, leukemia cells remained in his CSF. Although intrathecal alemtuzumab did not eliminate the CNS-invading leukemia cells, it was well-tolerated in our case. Further accumulation of similar cases is needed to determine the benefits and safety of intrathecal alemtuzumab administration.
T 细胞幼淋巴细胞白血病（T-PLL）是一种预后不佳的罕见血液系统癌症。尽管少数患者有中枢神经系统（CNS）受累，但尚未确立针对这些患者的标准治疗方法。在此，我们报告一例伴有 CNS 受累的 T-PLL 病例，其接受了比先前报道更高剂量的鞘内阿仑单抗治疗。一名患有伴有 CNS 受累的 T-PLL 的 66 岁男性入住我院。静脉注射阿仑单抗（一种抗 CD52 抗原的单克隆抗体）成功减少了外周血中的白血病细胞；然而，鞘内注射甲氨蝶呤、阿糖胞苷和泼尼松对 CNS 受累的治疗效果有限。因此，我们鞘内注射阿仑单抗作为实验性治疗。尽管我们将鞘内阿仑单抗的剂量逐步增加至 5mg，但未观察到不良反应；然而，该治疗并未完全清除患者脑脊液（CSF）中的白血病细胞。我们进行了全脑和全脊髓照射治疗，随后接受了来自人类白细胞抗原匹配的同胞供体的异基因移植，并采用了包含全身照射的预处理方案。移植后 21 天，其 CSF 中仍有白血病细胞。尽管鞘内阿仑单抗未能消除侵入 CNS 的白血病细胞，但在我们的病例中耐受性良好。需要进一步积累类似病例以确定鞘内注射阿仑单抗的益处和安全性。
