A case of natalizumab‐associated progressive multifocal leukoencephalopathy followed by immune reconstitution inflammatory syndrome with difficulty in the timing of immunotherapy
A case of natalizumab‐associated progressive multifocal leukoencephalopathy followed by immune reconstitution inflammatory syndrome with difficulty in the timing of immunotherapy
复制标题
那他珠单抗相关进行性多灶性白质脑病继发免疫重建炎症综合征且难以确定免疫治疗时机的一例
DOI：
10.1111/cen3.12734
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发表时间：
2022
影响因子：
--
通讯作者：
Maruyama Hirofumi
中科院分区：
文献类型：
--
作者：
Sugimoto Takamichi;Neshige Shuichiro;Aoki Shiro;Ochi Kazuhide;Ishikawa Ruoyi;Nonaka Megumi;Nakamori Masahiro;Nezu Tomohisa;Nakamichi Kazuo;Yamazaki Yu;Maruyama Hirofumi
背景尚未在日本患者中报告关于那他珠单抗相关的进行性多灶性白质脑病（NAT-PML）的临床过程的详细信息。我们经历了一个日本NAT-PML的情况下，并报告它的目的是澄清它在医疗带来的挑战。病例介绍在此，我们描述了一个58岁的多发性硬化症患者谁有NAT-PML免疫重建炎症综合征（IRIS）。在NAT-PML发生之前，患者的扩展残疾状态量表（EDSS）评分为5.5。她每3-7周接受一次那他珠单抗（300 mg），她的EDSS评分在9.1年内没有变化。在NAT-PML发生之前，John Cunningham病毒（JCV）指数为0.43。当NAT-PML出现时，她出现步态障碍。那他珠单抗共给药108次。基于T2加权成像上的点状病变和脑脊液（CSF）检测的JCV-PCR结果为29拷贝/ml，患者被诊断为可能的NAT-PML。当首次检测到造影剂增强病变时，开始静脉注射甲基强的松龙（IVMP），但尽管造影剂增强病变暂时消失，但NAT‐PML病变仍相当扩大。在免疫应答被激活时，IgG指数明显增加，CSF细胞计数略有增加。IVMP治疗IRIS每2周1次，疗程3个周期，EDSS评分最差为9.5分。结论IVMP治疗应在NAT-PML发病前进行。CSF细胞计数和IgG指数的变化可能有助于治疗决策;需要进一步研究。
BackgroundDetails regarding the clinical course of natalizumab‐associated progressive multifocal leukoencephalopathy (NAT‐PML) have not been reported in Japanese patients. We experienced a Japanese NAT‐PML case and report it for the purpose of clarifying the challenge it posed in medical treatment.Case PresentationHerein, we describe a 58‐y‐old multiple sclerosis patient who had NAT‐PML with immune reconstitution inflammatory syndrome (IRIS). Before NAT‐PML developed, the patient's Expanded Disability Status Scale (EDSS) score was 5.5. She received natalizumab (300 mg) every 3–7 w, and her EDSS score had not changed for 9.1 y. The John Cunningham virus (JCV) index was 0.43 before NAT‐PML developed. She developed a gait disturbance when NAT‐PML emerged. Natalizumab was administered a total of 108 times. The patient was diagnosed with probable NAT‐PML on the basis of punctate lesions on T2‐weighted imaging and a JCV‐PCR result of 29 copies/ml from cerebrospinal fluid (CSF) testing. Intravenous methylprednisolone (IVMP) was initiated when the contrast‐enhanced lesion was first detected, but the NAT‐PML lesion was rather enlarged despite the temporary disappearance of the contrast‐enhanced lesion. An obvious increase in the IgG index and a slight increase in the CSF cell count were recognized at the time the immunological response was activated. Three cycles of a 3‐d course of IVMP were administered every 2 w for IRIS, and the patient's worst EDSS score was 9.5.ConclusionTreatment sequencing should be executed before the onset of NAT‐PML. Changes in CSF cell count and IgG index may be useful for treatment decision; further research is needed.
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影响因子：
9.9
作者：
Landi, Doriana;De Rossi, Nicola;Centonze, Diego
通讯作者：
Centonze, Diego
影响因子：
9.9
作者：
Plavina T;Muralidharan KK;Kuesters G;Mikol D;Evans K;Subramanyam M;Nestorov I;Chen Y;Dong Q;Ho PR;Amarante D;Adams A;De Sèze J;Fox R;Gold R;Jeffery D;Kappos L;Montalban X;Weinstock-Guttman B;Hartung HP;Cree BAC
通讯作者：
Cree BAC
影响因子：
3.7
作者：
Cadavid D;Jurgensen S;Lee S
通讯作者：
Lee S
教师意见建议点状模式：用于诊断那他珠单抗相关 PML 的有前途的成像标记物。
DOI：
10.3410/f.726233577.793518661
发表时间：
2016
期刊：
影响因子：
--
作者：
S. Cook
通讯作者：
S. Cook
影响因子：
11
作者：
Wattjes, Mike P.;Wijburg, Martijn T.;Killestein, Joep
通讯作者：
Killestein, Joep