Weingertner Noëlle, Mitcov Mona, Chenard Marie-Pierre, Cribier Bernard
Department of Pathology, Strasbourg University Hospital, Strasbourg, France.
Department of Dermatology, Strasbourg University Hospital, Strasbourg, France.
J Cutan Pathol. 2016 Nov;43(11):1036-1040. doi: 10.1111/cup.12772. Epub 2016 Aug 22.
We describe the case of a patient presenting with drug rash with eosinophilia and systemic symptoms (DRESS), where cutaneous biopsy revealed intravascular atypical lymphocytes suggestive of lymphoma.
A 77-year-old man was treated with antibiotics for a hip prosthesis infection. Eight weeks later, he developed a maculo-papulous eruption, edema of the extremities, fever and blood eosinophilia. Cutaneous biopsy revealed an atypical T-cell proliferation into the dermal lymphatic vessels. The lymphocytes were mid-sized, with mitoses and apoptotic figures. They were CD3+, CD4+, CD5+ and some were CD30+. There was no T-cell receptor (TcR) clonal rearrangement. Complete regression of cutaneous eruption and eosinophilia was observed after ceasing treatment with antibiotics. The diagnosis was that of a benign atypical intralymphatic T-cell proliferation associated with DRESS.
The occurrence of atypical dermal CD30+ T-cells in cutaneous biopsy during benign reactive conditions such as arthropod bites or scabies is well-known. The intralymphatic localization of such atypical reactive lymphocytes is much less common and represents a diagnostic pitfall because it can suggest aggressive intravascular lymphoma.
We report the first case of benign atypical intralymphatic CD30+ T-cell proliferation associated with DRESS. Diagnostic clues include immunohistochemistry, absence of TcR clonal rearrangement, and anatomo-clinical correlation.
我们描述了一例出现药物疹伴嗜酸性粒细胞增多和全身症状(DRESS)的患者,其皮肤活检显示血管内非典型淋巴细胞,提示淋巴瘤。
一名77岁男性因髋关节假体感染接受抗生素治疗。八周后,他出现了斑丘疹、四肢水肿、发热和血液嗜酸性粒细胞增多。皮肤活检显示真皮淋巴管内有非典型T细胞增殖。淋巴细胞中等大小,有有丝分裂和凋亡小体。它们CD3+、CD4+、CD5+,部分为CD30+。未发现T细胞受体(TcR)克隆重排。停用抗生素后,皮肤疹和嗜酸性粒细胞增多完全消退。诊断为与DRESS相关的良性非典型淋巴管内T细胞增殖。
在良性反应性疾病如节肢动物叮咬或疥疮时,皮肤活检中出现非典型真皮CD30+ T细胞是众所周知的。这种非典型反应性淋巴细胞的淋巴管内定位则少见得多,是一个诊断陷阱,因为它可能提示侵袭性血管内淋巴瘤。
我们报告了首例与DRESS相关的良性非典型淋巴管内CD30+ T细胞增殖病例。诊断线索包括免疫组化、TcR克隆重排阴性以及解剖学与临床的相关性。