Pawson R, Matutes E, Brito-Babapulle V, Maljaie H, Hedges M, Mercieca J, Dyer M, Catovsky D
Academic Department of Haematology and Cytogenetics, Royal Marsden Hospital, London, UK.
Leukemia. 1997 Jul;11(7):1009-13. doi: 10.1038/sj.leu.2400710.
We report the clinical, ultrastructural, immunophenotypic and virological features of nine cases of a rare type of mature T cell disorder formerly designated Sezary cell leukaemia. All patients presented with lymphocytosis ranging from 12.7 to 133 x 10(9)/l, bone marrow infiltration, splenomegaly and lymphadenopathy. Skin involvement was absent at presentation but developed as a terminal event in two patients, one of whom showed a pattern of dermal infiltration different from that characteristic of Sezary syndrome. Cells from eight cases bore a mature T cell phenotype and electronmicroscopy revealed lymphocytes with cerebriform nuclei resembling Sezary cells. All cases except one were HTLV-I negative. Patients were treated with various chemotherapy regimens but with poor outcome, the median survival being 13 months. Laboratory and clinical data suggest great similarity between Sezary cell leukaemia and T prolymphocytic leukaemia (T-PLL), namely coexpression of CD4 and CD8 (3/9 cases), identical chromosomal abnormalities in the three cases studied (isochromosome 8q plus inversion 14 or t(X;14)(q28;q11)) and a remarkable sensitivity to CAMPATH-1H (complete remission of 21 months' duration in one patient), suggesting that this entity could be considered a variant form of T-PLL. The alternative diagnosis of adult T cell leukaemia/lymphoma could not be excluded in one patient in whom positive HTLV-I serology was documented.
我们报告了9例罕见类型成熟T细胞疾病(以前称为Sezary细胞白血病)的临床、超微结构、免疫表型和病毒学特征。所有患者均表现为淋巴细胞增多,范围为12.7至133×10⁹/L,有骨髓浸润、脾肿大和淋巴结病。初诊时无皮肤受累,但有2例患者在终末期出现皮肤受累,其中1例患者的皮肤浸润模式与Sezary综合征的特征不同。8例患者的细胞具有成熟T细胞表型,电子显微镜检查显示淋巴细胞有脑回状核,类似于Sezary细胞。除1例患者外,所有病例HTLV-I均为阴性。患者接受了各种化疗方案,但预后较差,中位生存期为13个月。实验室和临床数据表明,Sezary细胞白血病与T原淋巴细胞白血病(T-PLL)极为相似,即CD4和CD8共表达(3/9例),在研究的3例患者中染色体异常相同(8号染色体长臂等臂染色体加14号染色体倒位或t(X;14)(q28;q11)),且对CAMPATH-1H具有显著敏感性(1例患者完全缓解持续21个月),这表明该实体可被视为T-PLL的一种变异形式。1例记录有HTLV-I血清学阳性的患者不能排除成人T细胞白血病/淋巴瘤的鉴别诊断。