Yamanaka Satoshi, Nakayama Kazutaka, Tamai Hayato, Sakamaki Masanori, Inokuchi Koiti
Department of Hematology, Nippon Medical School.
Department of Neurology, Nippon Medical School.
Rinsho Ketsueki. 2017;58(4):309-314. doi: 10.11406/rinketsu.58.309.
Adult T-cell leukemia-lymphoma (ATL) caused by human T-lymphotropic virus type 1 (HTLV-1) is generally associated with poor prognosis. The anti-CCR4 antibody mogamulizumab is one of the options for refractory or relapsed ATL. Mogamulizumab is intravenously administered as a single agent at a dose of 1.0 mg/kg once a week for 8 weeks. The overall response rate is reported to be 50%. Reported common adverse events after administration of mogamulizumab are leukocytopenia, infusion reaction, and skin rash. Although rare, mogamulizumab-associated cardiomyopathy has also been reported. HTLV-1 is also associated with a number of diseases, such as HTLV-1-associated myelopathy (HAM). Approximately 0.25% of infected individuals are estimated to develop HAM. HAM is an inflammatory disease of the central nervous system, which is characterized by slowly progressive spastic paresis of bilateral lower limbs and bladder-rectal disorder. Coexistence of ATL and HAM has rarely been reported. Here we present the case of a patient with ATL in whom Takotsubo cardiomyopathy and HAM developed after mogamulizumab administration.
由1型人类嗜T淋巴细胞病毒（HTLV-1）引起的成人T细胞白血病-淋巴瘤（ATL）通常预后较差。抗CCR4抗体莫加莫单抗是难治性或复发性ATL的治疗选择之一。莫加莫单抗作为单一药物静脉给药，剂量为1.0 mg/kg，每周一次，共8周。据报道，总体缓解率为50%。据报道，使用莫加莫单抗后常见的不良事件有白细胞减少、输液反应和皮疹。虽然罕见，但也有莫加莫单抗相关心肌病的报道。HTLV-1还与多种疾病有关，如HTLV-1相关脊髓病（HAM）。据估计，约0.25%的感染者会发展为HAM。HAM是一种中枢神经系统炎症性疾病，其特征是双侧下肢缓慢进行性痉挛性麻痹和膀胱直肠功能障碍。ATL和HAM并存的情况鲜有报道。在此，我们报告一例ATL患者，在使用莫加莫单抗后发生了Takotsubo心肌病和HAM。
