Primary cutaneous anaplastic large cell lymphoma with fatal leukemic outcome in association with CLA and CCR4-negative conversion

Primary cutaneous anaplastic large cell lymphoma with fatal leukemic outcome in association with CLA and CCR4-negative conversion
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DOI:
10.1016/j.jaad.2006.08.053
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发表时间:
2007-11-01
影响因子:
13.8
通讯作者:
Takigawa, Masahiro
Takigawa, Masahiro
中科院分区:
医学1区
文献类型:
--
作者:
Tokura, Yoshiki;Sugita, Kazunari;Takigawa, Masahiro

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一名70岁的日本男性,有1年的皮肤肿瘤病史,由于CD 3(低+)、CD+、CD 25+、CD 30+、CD 45 RO(+)、CD 71(+)、HLA-DR+、CD 8(-)、CD 56(-)、和NPM/ALK(-)表型和肿瘤细胞的单克隆T细胞受体重排特性以及不存在全身性受累。此时,肿瘤细胞对皮肤淋巴细胞相关抗原(CLA)和TH 2趋化因子受体CCR 4呈阳性。出疹反复出现,并通过多种治疗方式(包括放疗、干扰素-a和化疗)自发消退或消退,直至5年后肿瘤细胞侵入胃粘膜并扩散至外周血。在进展为致死性白血病变化时,皮肤病变反向消失。流式细胞仪检测外周血和皮肤浸润淋巴细胞表型显示,肿瘤细胞上CIA和CCR 4的表达由阳性转为阴性,与白血病的变化有关。皮肤归巢受体表达的改变可能会改变其临床行为。
A 70-year-old Japanese male presented with a 1-year history of skin tumors, which were diagnosed as primary cutaneous anaplastic large cell lymphoma (ALCL) because of the CD3(low +), CD+, CD25+, CD30+, CD45RO(+), CD71(+), HLA-DR+, CD8(-), CD56(-), and NPM/ALK(-) phenotype and monoclonal T-cell receptor-rearranged property of tumor cells as well as the absence of systemic involvement. At this time, the tumor cell was positive for cutaneous lymphocyte-associated antigen (CLA) and TH2 chemokine receptor CCR4. The eruption had repeatedly appeared and spontaneously regressed or regressed by virtue of several therapeutic modalities, including radiotherapy, interferon-a and chemotherapy, until the tumor cell invaded the gastric mucosa and spread to the peripheral blood 5 years later. Upon progression to the fatal leukemic change, the skin lesions inversely disappeared. Flow cytometric monitoring of the phenotype of peripheral blood and skin-infiltrating lymphocytes disclosed that the expression of CIA and CCR4 on the tumor cells was converted from positive to negative in association with the leukemic change. The altered expression of skin-homing receptors might change its clinical behavior.