Wain E Mary, Orchard Guy E, Mayou Susan, Atherton David J, Misch Klaus J, Russell-Jones Robin
Skin Tumor Unit, St John's Institute of Dermatology, St Thomas' Hospital, London, UK.
J Am Acad Dermatol. 2005 Jul;53(1):158-63. doi: 10.1016/j.jaad.2005.01.133.
We report 3 cases of mycosis fungoides (MF) with a CD56+ cytotoxic immunophenotype. Each patient presented with a different clinical phenotype: one exhibited limited poikilodermatous patches (skin stage T1); one, widespread hypopigmented lesions (skin stage T2); and one, poikiloderma with a single cutaneous tumor (skin stage T3). MF was confirmed both histologically and by the presence of a T-cell receptor clone in lesional skin in all cases. CD56 and T-cell intracellular antigen-1 were expressed by the malignant lymphocytes in all patients and two expressed CD8. No sample demonstrated loss of the pan T-cell markers CD2 or CD3. None of the 3 developed systemic disease and T-cell receptor gene analysis of peripheral blood was polyclonal in all cases. Only 3 cases of CD56+ MF have been reported previously, none of which exhibited tumor-stage disease. Currently, the disease in our patients appears to be behaving in a manner similar to that predicted for MF with a normal immunophenotype but the prognosis has to be guarded in view of the rarity of this subtype.
我们报告了3例具有CD56 + 细胞毒性免疫表型的蕈样肉芽肿(MF)病例。每位患者表现出不同的临床表型:一位表现为局限性斑驳样皮疹(皮肤T1期);一位表现为广泛的色素减退性损害(皮肤T2期);另一位表现为伴有单个皮肤肿瘤的斑驳样皮疹(皮肤T3期)。所有病例经组织学检查及病变皮肤中T细胞受体克隆的存在均确诊为MF。所有患者的恶性淋巴细胞均表达CD56和T细胞胞内抗原-1,其中两位表达CD8。没有样本显示泛T细胞标志物CD2或CD3缺失。3例患者均未发展为系统性疾病,所有病例外周血T细胞受体基因分析均为多克隆性。此前仅报道过3例CD56 + MF,均未表现出肿瘤期疾病。目前,我们患者的疾病表现似乎与具有正常免疫表型的MF预期表现相似,但鉴于该亚型罕见,预后仍需谨慎。