Zhang Ling, Ramchandren Radhakrishnan, Papenhausen Peter, Loughran Thomas P, Sokol Lubomir
Department of Hematopathology and Laboratory Medicine, H Lee Moffitt Cancer Center and Research Institute, Tampa, FL, USA.
Eur J Haematol. 2014 Sep;93(3):260-4. doi: 10.1111/ejh.12313. Epub 2014 Apr 7.
T-cell large granular lymphocytic leukemia (T-LGLL) is a rare indolent lymphoproliferative disorder characterized by cytopenias, splenomegaly, and various degrees of T-cell lymphocytosis, due to a clonal expansion of CD8-positive cytotoxic T-cells. Phenotypic variants of T-LGLL include CD4(+) /CD8(-) T-cells, with dual CD4(-) /CD8(-) /γδ(+) T-cells being even rarer. Cytogenetic abnormalities in T-LGLL have rarely been reported, and there is scientific debate regarding the existence of aggressive or transformed variants of T-LGLL. We report a patient with T-LGLL, γδ variant, with nearly 20-year-long duration of cytopenias before transformation to an unusual clinical scenario, manifesting with marked lymphocytosis >100 × 10(9) /L and infiltration of lymph nodes, tonsils, and subcutaneous tissue. Single-nucleotide polymorphism assays revealed acquired copy neutral loss of heterozygosity at 17q and deletion of 3p21.31, in addition to trisomy 5, monosomy X, and monosomy 21. These genetic abnormalities provided a better understanding of the molecular nature and the potentiality of disease transformation.
T 细胞大颗粒淋巴细胞白血病(T-LGLL)是一种罕见的惰性淋巴细胞增殖性疾病,其特征为血细胞减少、脾肿大以及不同程度的 T 细胞淋巴细胞增多,这是由于 CD8 阳性细胞毒性 T 细胞的克隆性扩增所致。T-LGLL 的表型变异包括 CD4(+) /CD8(-) T 细胞,双阴性 CD4(-) /CD8(-) /γδ(+) T 细胞则更为罕见。T-LGLL 的细胞遗传学异常鲜有报道,关于 T-LGLL 是否存在侵袭性或转化型变异也存在科学争议。我们报告了 1 例γδ变异型 T-LGLL 患者,在转变为一种不寻常的临床情况之前,血细胞减少持续了近 20 年,表现为显著的淋巴细胞增多>100×10⁹/L,以及淋巴结、扁桃体和皮下组织浸润。单核苷酸多态性分析显示,除了 5 号染色体三体、X 染色体单体和 21 号染色体单体外,还存在位于 17q 的获得性拷贝数中性杂合性缺失以及 3p21.31 缺失。这些基因异常有助于更好地理解该疾病的分子本质以及疾病转化的可能性。