Uchida Tomoyuki, Inoue Morihiro, Hua Jian, Tajima Shogo, Ota Yasunori, Hagihara Masao
Department of Hematology, Eiju General Hospital.
Department of Pathology, Eiju General Hospital.
Rinsho Ketsueki. 2017;58(6):624-629. doi: 10.11406/rinketsu.58.624.
Here we present a patient with rheumatoid arthritis (RA), who was suspected to have developed malignant lymphoma during immunosuppressive therapy 5 years earlier. She temporarily achieved remission after discontinuing therapy; however, her disease worsened with remittent fever and splenomegaly. Splenic biopsy demonstrated infiltration by abnormal cells, which were positive for CD56 and T cell intracytoplasmic antigen, but negative for CD3 and Epstein-Barr virus (EBV) -encoded RNA. Cytogenetic analysis of bone marrow and lumbar spine tumor revealed common complex karyotype abnormalities. Thus, she was diagnosed with chronic natural killer cell lymphoproliferative disorder (NK-LPD) and finally died of disease progression. The most common type of LPD in methotrexate-related patients with RA is B-lymphoid LPD, whereas NK-LPD is extremely rare. Furthermore, almost all cases of NK-LPD have been reported to be positive for EBV. This is the first case report on a patient with EBV-negative NK-LPD developed during immunosuppressive therapy for RA.
在此,我们报告一名类风湿关节炎(RA)患者,该患者在5年前接受免疫抑制治疗期间疑似发生恶性淋巴瘤。停药后她暂时实现缓解;然而,她的病情因弛张热和脾肿大而恶化。脾脏活检显示有异常细胞浸润,这些细胞CD56和T细胞胞浆内抗原呈阳性,但CD3和EB病毒(EBV)编码的RNA呈阴性。骨髓和腰椎肿瘤的细胞遗传学分析显示存在常见的复杂核型异常。因此,她被诊断为慢性自然杀伤细胞淋巴增殖性疾病(NK-LPD),最终死于疾病进展。在与甲氨蝶呤相关的RA患者中,最常见的LPD类型是B淋巴细胞LPD,而NK-LPD极为罕见。此外,几乎所有NK-LPD病例据报道EBV均呈阳性。这是首例关于在RA免疫抑制治疗期间发生EBV阴性NK-LPD患者的病例报告。