Kinsella Francesca A M, Amel Kashipaz Mohammad Rasoul, Scarisbrick Julia, Malladi Ram
Institute of Immunology and Immunotherapy, University of Birmingham, Birmingham, UK.
Department of Cellular Pathology, University Hospital Birmingham NHS Foundation Trust, Birmingham, UK.
BMJ Case Rep. 2017 Jan 10;2017:bcr2016216331. doi: 10.1136/bcr-2016-216331.
A 46-year-old woman with a history of dasatinib-resistant chronic myeloid leukaemia, clonal evolution and monosomy 7 underwent reduced intensity conditioned in vivo T-cell-depleted allogeneic haematopoietic stem cell transplantation (HSCT) from a matched unrelated donor. Following the transplantation, she developed recurrent cutaneous graft versus host disease (GvHD), which required treatment with systemic immunosuppression and electrocorporeal photophoresis. Concurrently, she developed a lichenoid rash with granulomatous features suggestive of cutaneous sarcoidosis. Additional treatment with hydroxychloroquine was initially successful, but 2 months later, she developed erythroderma with palpable lymphadenopathy. Repeated histological analysis established a diagnosis of folliculotropic mycosis fungoides stage IVA2, and the malignant clone was confirmed to be of donor origin. A positive response to brentuximab has been shown. This is the first reported case of primary mycosis fungoides after matched unrelated donor HSCT, and in a patient still undergoing treatment for GvHD.
一名46岁女性，有达沙替尼耐药的慢性髓性白血病、克隆进化和7号染色体单体病史，接受了来自匹配无关供体的低强度预处理体内T细胞清除的异基因造血干细胞移植（HSCT）。移植后，她出现了复发性皮肤移植物抗宿主病（GvHD），需要全身免疫抑制和体外光化学疗法治疗。同时，她出现了具有肉芽肿特征的苔藓样皮疹，提示皮肤结节病。最初使用羟氯喹进行额外治疗取得了成功，但2个月后，她出现了红皮病并伴有可触及的淋巴结病。反复的组织学分析确诊为毛囊性蕈样肉芽肿IVA2期，且恶性克隆被证实来自供体。已显示对brentuximab有阳性反应。这是首例报道的匹配无关供体HSCT后发生的原发性蕈样肉芽肿病例，且该患者仍在接受GvHD治疗。
