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病例报告:多发性硬化症患者在接受第二次阿仑单抗治疗后出现肿块样脱髓鞘病变;免疫细胞特征和生物标志物。

Case report: tumefactive demyelinating lesions after the second cycle of alemtuzumab in multiple sclerosis; immune cell profile and biomarkers.

机构信息

Neurosciences Department, Hospital Universitari Germans Trias i Pujol, Badalona, Spain.

Immunology Division, Clinical Laboratory MetroNord (LCMN), Hospital Universitari Germans Trias i Pujol and Research Institute (IGTP), Badalona, Spain.

出版信息

Front Immunol. 2024 Jul 3;15:1395749. doi: 10.3389/fimmu.2024.1395749. eCollection 2024.

DOI:10.3389/fimmu.2024.1395749
PMID:39021563
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11251942/
Abstract

OBJECTIVE

We present a case of multiple tumefactive demyelinating lesions (TDLs) emerging 24 months after the second cycle of alemtuzumab treatment.

METHODS

A woman with relapsing-remitting multiple sclerosis (MS) discontinued fingolimod treatment due to gestational desire, which resulted in a severe disease exacerbation. Alemtuzumab was initiated, accompanied by regular clinical, radiological, and immunological monitoring.

RESULTS

She relapsed prior to the second cycle, exhibiting 12 T1Gd lesions, and peripheral blood showed an increase in B-cells and a decrease in T-cells. At 24 months following the second cycle, she developed cognitive impairment and multiple T1Gd lesions, including TDLs, were evident on the brain MRI. We found not only an increase in B-cells but also in Th1 central memory cells. Th1/Th17 cells increased 3 months before the detection of TDLs.

CONCLUSIONS

TDLs can appear 24 months after the second cycle of alemtuzumab treatment in MS. The increase in Th1/Th17 cells could be a candidate biomarker for TDLs in alemtuzumab-treated MS patients.

摘要

目的

我们报告 1 例多发性肿块性脱髓鞘病变(TDL),出现在第 2 周期阿仑单抗治疗后 24 个月。

方法

1 例复发性缓解型多发性硬化症(MS)患者因妊娠需求停用芬戈莫德治疗,导致疾病严重恶化。给予阿仑单抗治疗,并进行定期的临床、影像学和免疫学监测。

结果

她在第 2 周期前复发,出现 12 个 T1Gd 病变,外周血显示 B 细胞增加,T 细胞减少。第 2 周期后 24 个月,她出现认知障碍,脑 MRI 显示多个 T1Gd 病变,包括 TDL。我们不仅发现 B 细胞增加,还发现 Th1 中央记忆细胞增加。TDLs 出现前 3 个月,Th1/Th17 细胞增加。

结论

MS 患者在接受第 2 周期阿仑单抗治疗后 24 个月可能会出现 TDL。Th1/Th17 细胞的增加可能是阿仑单抗治疗 MS 患者 TDL 的候选生物标志物。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/47de0386b9ab/fimmu-15-1395749-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/ba22fa7a753c/fimmu-15-1395749-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/a4e1558c6589/fimmu-15-1395749-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/47de0386b9ab/fimmu-15-1395749-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/ba22fa7a753c/fimmu-15-1395749-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/a4e1558c6589/fimmu-15-1395749-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa57/11251942/47de0386b9ab/fimmu-15-1395749-g003.jpg

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本文引用的文献

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GABA receptor autoimmunity after alemtuzumab treatment for multiple sclerosis.阿仑单抗治疗多发性硬化症后出现的GABA受体自身免疫反应。
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Stabilization Without Rituximab After Disease Activation in an Alemtuzumab-Treated Patient with Multiple Sclerosis and a Literature Overview.一名接受阿仑单抗治疗的多发性硬化症患者疾病激活后未使用利妥昔单抗的病情稳定情况及文献综述
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Unexpected high multiple sclerosis activity after switching from fingolimod to alemtuzumab.从芬戈莫德转换为阿仑单抗后出现意外的多发性硬化症活动。
Mult Scler Relat Disord. 2018 Oct;25:216-218. doi: 10.1016/j.msard.2018.08.006. Epub 2018 Aug 7.
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Multiple sclerosis: severe exacerbation after stopping disease-modifying drugs.多发性硬化症:停用疾病修正药物后严重恶化。
Eur J Neurol. 2018 Oct;25(10):1199-1200. doi: 10.1111/ene.13746. Epub 2018 Jul 20.
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Activation of disease during therapy with alemtuzumab in 3 patients with multiple sclerosis.3 例多发性硬化症患者在用阿仑单抗治疗期间疾病激活。
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Evidence of B-cell dysregulation in severe CNS inflammation after alemtuzumab therapy.阿仑单抗治疗后严重中枢神经系统炎症中B细胞失调的证据。
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