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与 Brentuximab Vedotin 治疗相关的 PRES 样白质脑病伴癫痫持续状态。

PRES-like leukoencephalopathy presenting with status epilepticus associated with Brentuximab Vedotin treatment.

机构信息

Pediatric Neurology, Pediatric University Department, Azienda Ospedaliera Universitaria Pisana, University of Pisa, Italy.

Pediatric Neurology Unit, Dinogmi, Giannina Gaslini's Istitute, University of Genoa, Italy.

出版信息

Acta Biomed. 2022 Mar 21;92(S4):e2021416. doi: 10.23750/abm.v92iS4.12665.

Abstract

Posterior Reversible Encephalopathy Syndrome (PRES) is characterized by acute neurological symptoms with typical imaging features, primarily in the territories of the brain supplied by the posterior circulation, probably due to vasogenic edema. Both clinical and imaging features are generally reversible. We report a 13-year-old girl affected by Nodular Sclerosis Classical Hodgkin Lymphoma stage IIIB into complete remission, with a recurrence and autologous bone-marrow transplantation, who has been treated with an anti-CD30 monoclonal antibody, brentuximab-vedotin. The girl has suddenly presented a convulsive status epilepticus, that needed intubation and sedation. Therefore, an IV therapy with levetiracetam was started. Furthermore, the girl has presented high blood pressure and reduced kidney function. Brain MRI demonstrated a diffuse PRES-like disease, that went into regression after the first week. After another week, the girl presented a new prolonged generalized tonic clonic convulsive episode, that needed intubation and sedation and an association of clobazam and levetiracetam: a new brain MRI showed a recurrence of PRES-like lesions in addition to some signs of leukoencephalopathy with brain lactate accumulation on 1H-MRS, due to cerebral energetic failure. The girl also presented a refractory arterial hypertension. After 45 days of ICU hospitalization the patient has been discharged and followed up with neurological examinations. Brain MRI and brain 1H-MRS, 5 months after patient's discharge, showed incomplete regression of cerebral white matter signal abnormalities with MRS normalization.

摘要

后部可逆性脑病综合征(PRES)的特点是急性神经系统症状伴有典型的影像学特征,主要发生在大脑后循环供血区,可能与血管源性水肿有关。临床和影像学特征通常是可逆的。我们报告了一例 13 岁女孩,患有结节性硬化型经典霍奇金淋巴瘤 IIIB 期,完全缓解后复发并接受自体骨髓移植,接受了抗 CD30 单克隆抗体 Brentuximab-Vedotin 治疗。该女孩突然出现癫痫持续状态,需要插管和镇静。因此,开始给予静脉注射左乙拉西坦治疗。此外,该女孩还出现高血压和肾功能减退。脑部 MRI 显示弥漫性 PRES 样疾病,在第一周后开始消退。一周后,该女孩又出现新的长时间全身强直阵挛性癫痫发作,需要插管和镇静,并联合使用氯巴占和左乙拉西坦:新的脑部 MRI 显示 PRES 样病变复发,此外,1H-MRS 显示脑乳酸堆积的脑白质病变,提示脑能量衰竭。该女孩还出现了难治性动脉高血压。在 ICU 住院 45 天后,患者出院并进行了神经学检查。患者出院后 5 个月进行脑部 MRI 和脑部 1H-MRS 检查,显示脑白质信号异常的不完全消退,MRS 正常化。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b20e/9179058/94a7994ed661/ACTA-92-416-g001.jpg

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