Gessain A, Moulonguet I, Flageul B, Perrin P, Capesius C, D'Agay M F, Gisselbrecht C, Sigaux F, Civatte J
Laboratorie d'Hématologie Moléculaire, Hôpital Saint-Louis, Paris France.
J Am Acad Dermatol. 1990 Nov;23(5 Pt 2):994-1000. doi: 10.1016/0190-9622(90)70321-8.
A 45-year-old woman, a native of the French West Indies who had lived in France since 1973, developed multiple cutaneous plaques and nodules in 1987. Histopathologic studies revealed dermal infiltration with mature activated T cells (CD4+, CD25+, DR+) with nuclear convolutions and epidermatotropisim. High titers of specific human T lymphotropic virus (HTLV)-I antibodies were detected in the serum. Molecular analysis of DNA extracted from the skin tumor biopsy specimen showed a clonal integration of an HTLV-I provirus and a T-cell clonal population as demonstrated by T-cell receptor beta and gamma gene rearrangement studies. Neither HTLV-I provirus nor T-cell receptor rearrangements were detected in peripheral blood mononuclear cells DNA despite the presence of rare adult T cell leukemia cells (less than 1%) and a small excess of DR-expressing cells, and detection of HTLV-I Pol and Px sequences by in vitro gene amplification. In this case only gene analysis of the skin lesions made possible an early diagnosis of a cutaneous adult T cell leukemia. This illustrates the need for such molecular studies to differentiate, in HTLV-I seropositive patients from endemic areas, a HTLV-I-induced T cell lymphoma from HTLV-I-nonrelated cutaneous T cell lymphomas.
一名45岁女性,出生于法属西印度群岛,自1973年起居住在法国,1987年出现多处皮肤斑块和结节。组织病理学研究显示真皮内有成熟的活化T细胞(CD4 +、CD25 +、DR +)浸润,伴有核卷曲和向表皮性。血清中检测到高滴度的特异性人类嗜T淋巴细胞病毒(HTLV)-I抗体。从皮肤肿瘤活检标本中提取的DNA分子分析显示,HTLV-I前病毒存在克隆整合以及T细胞克隆群体,这通过T细胞受体β和γ基因重排研究得以证实。尽管存在罕见的成人T细胞白血病细胞(少于1%)和少量过量表达DR的细胞,并且通过体外基因扩增检测到HTLV-I Pol和Px序列,但在外周血单个核细胞DNA中未检测到HTLV-I前病毒和T细胞受体重排。在这种情况下,仅对皮肤病变进行基因分析才得以早期诊断皮肤型成人T细胞白血病。这说明了对于来自流行地区的HTLV-I血清阳性患者,进行此类分子研究以区分HTLV-I诱导的T细胞淋巴瘤与HTLV-I无关的皮肤T细胞淋巴瘤的必要性。