Donor-derived mycosis fungoides following reduced intensity haematopoietic stem cell transplantation from a matched unrelated donor.
Donor-derived mycosis fungoides following reduced intensity haematopoietic stem cell transplantation from a matched unrelated donor.
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供体衍生的霉菌病真菌病在降低匹配的无关供体的强度造血干细胞移植后。
DOI：
10.1136/bcr-2016-216331
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发表时间：
2017-01-10
期刊：
影响因子：
0.9
通讯作者：
Malladi R
中科院分区：
文献类型：
--
作者：
Kinsella FA;Amel Kashipaz MR;Scarisbrick J;Malladi R
一位46岁的女性，既有达沙替尼耐药的慢性髓系白血病病史，有克隆性进化和7号单体，接受了来自匹配的非血缘供者的体内T细胞耗竭异基因造血干细胞移植(HSCT)。移植后，她出现了反复发作的皮肤移植物抗宿主病(GvHD)，需要全身免疫抑制和电身体光导入治疗。同时，她出现了苔藓样皮疹，肉芽肿样特征提示皮肤结节病。额外的羟氯喹治疗最初是成功的，但2个 月后，她出现了红皮病和明显的淋巴结病。重复的组织学分析确定诊断为IVA2期的嗜毛囊性真菌病，恶性克隆被确认为供体起源。对布妥昔单抗的反应是积极的。这是第一例在匹配的非血缘关系供者HSCT后和仍在接受GvHD治疗的患者中报告的原发真菌病病例。
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.