Department of Internal Medicine, Seoul St. Mary's Hematology Hospital, College of Medicine, The Catholic University of Korea, Banpo-daero 222, Seocho-Gu, Seoul, 06591, Republic of Korea.
Leukemia Research Institute, College of Medicine, The Catholic University of Korea, Seoul, Korea.
Int J Hematol. 2019 Nov;110(5):635-639. doi: 10.1007/s12185-019-02702-1. Epub 2019 Jul 15.
Graft-versus-host disease (GVHD) is a serious complication of allogeneic stem cell transplantation (SCT). Here, we report a rare case of GVHD involving the central nervous system (CNS). A 35-year-old woman was diagnosed with myelodysplastic syndrome unclassifiable and underwent allogeneic peripheral blood SCT for disease progression to myelodysplastic syndrome with excess blasts-2. One year following SCT, she experienced chronic oral and hepatic GVHD symptoms, which were managed with oral steroids and tacrolimus. Sixteen months after SCT, she developed sudden-onset, generalized, tonic-clonic-type seizures. Magnetic resonance imaging and cerebrospinal fluid evaluation showed multiple discrete white lesions and elevated IgG levels. Brain biopsy revealed periventricular plaques with the destruction of axons, representing a demyelinating disease of the CNS. We diagnosed the case as CNS GVHD. Neurologic symptoms gradually improved with methylprednisolone pulse therapy and total plasma exchange combined with a calcineurin inhibitor; the brain lesions nearly disappeared after decreasing steroid maintenance dosage, and were completely resolved 1 year after the onset of CNS GVHD. The patient is CNS GVHD-symptom-free, 3-year post-transplantation. Thus, CNS GVHD should be considered in cases of newly developed neurologic symptoms in SCT recipients showing evidence of preceding chronic GVHD.
移植物抗宿主病(GVHD)是异基因造血干细胞移植(SCT)的严重并发症。在这里,我们报告了一例罕见的涉及中枢神经系统(CNS)的 GVHD 病例。一名 35 岁女性被诊断为非特指性骨髓增生异常综合征,并因疾病进展为伴原始细胞过多的骨髓增生异常综合征-2 而行异基因外周血 SCT。SCT 后 1 年,她出现慢性口腔和肝脏 GVHD 症状,经口服类固醇和他克莫司治疗。SCT 后 16 个月,她突发全身性强直阵挛性癫痫发作。磁共振成像和脑脊液评估显示多发性离散性白色病变和 IgG 水平升高。脑活检显示脑室周围斑块伴轴突破坏,代表中枢神经系统脱髓鞘疾病。我们诊断为 CNS GVHD。神经症状在甲基强的松龙脉冲治疗和总血浆置换联合钙调神经磷酸酶抑制剂后逐渐改善;降低类固醇维持剂量后脑损伤几乎消失,CNS GVHD 发病后 1 年完全消退。患者在移植后 3 年无 CNS GVHD 症状。因此,对于出现先前慢性 GVHD 证据的新发生神经系统症状的 SCT 受者,应考虑 CNS GVHD。