Machatschek J N, Westerman D, Prince H M, Seymour J F
Department of Haematology, Peter MacCallum Cancer Institute, St. Andrew's Place, East Melbourne, VIC 3002, Australia.
Leuk Lymphoma. 2003 Jul;44(7):1223-7. doi: 10.1080/1042819031000079203.
We report a case of natural killer (NK) cell leukemia with unusual biological features in a 65-year-old woman with a 20-year history of rheumatoid arthritis. She presented with neutropenia, thrombocytopenia, splenomegaly and bone marrow infiltration. Immunophenotyping (CD2+ CD3- CD4- CD5- CD8- CD16+ CD56-) confirmed NK cell leukemia. Her neutropenia and thrombocytopenia resolved following splenectomy and she has remained well with stable disease for 12 months on oral low-dose methotrexate. In contrast to all previous reports, in this instance the phenotype of large granular lymphocytic (LGL) leukemia occurring in the context of rheumatoid arthritis was NK-cell rather than T-cell. Furthermore, the clinical course was indolent, whereas, all prior literature reports have described a very aggressive clinical course for this disorder with a median survival of just 3.5 months. This case illustrates previously unrecognized heterogeneity in the natural history of this disorder.
我们报告了一例自然杀伤(NK)细胞白血病病例,该病例具有不寻常的生物学特征,患者为一名65岁女性,有20年类风湿关节炎病史。她表现为中性粒细胞减少、血小板减少、脾肿大和骨髓浸润。免疫表型分析(CD2 + CD3 - CD4 - CD5 - CD8 - CD16 + CD56 -)确诊为NK细胞白血病。脾切除术后她的中性粒细胞减少和血小板减少症状得到缓解,并且在口服低剂量甲氨蝶呤的情况下,病情稳定了12个月,情况良好。与之前所有报告不同的是,在本例中,发生在类风湿关节炎背景下的大颗粒淋巴细胞(LGL)白血病的表型是NK细胞而非T细胞。此外,临床病程呈惰性,而所有先前的文献报告都描述了这种疾病非常侵袭性的临床病程,中位生存期仅为3.5个月。该病例说明了这种疾病自然史中以前未被认识到的异质性。