Rituximab in Rasmussen's encephalitis: A single center experience and review of the literature.
Rituximab in Rasmussen's encephalitis: A single center experience and review of the literature.
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DOI：
10.1016/j.ebr.2022.100540
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发表时间：
2022
影响因子：
--
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中科院分区：
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在疾病早期使用利妥昔单抗可以很好地控制癫痫发作和EPC。利妥昔单抗可以逆转或稳定运动缺陷以及MRI变化，并阻止疾病进展。利妥昔单抗是一种安全、有效、耐受性好、作用迅速的药物，可作为类固醇保留剂使用。拉斯穆森脑炎（RE）是一种罕见的脑部慢性炎症性疾病，导致单侧半球萎缩并伴有耐药局灶性癫痫，并伴有不同程度的进行性偏瘫和认知能力下降。RE的确切病因尚不清楚，但推测有神经炎症病理生物学基础。只有手术才能阻止疾病的进展，但可能以固定但不可避免的神经功能缺损为代价。因此，医疗管理问题是一个重要的考虑因素。2008年美国癫痫学会年会上发表了关于在RE患者中使用利妥昔单抗的报告。从那时起，关于它的使用的良好的公开证据就非常缓慢地出现。然而，利妥昔单抗继续被列在RE患者治疗方案的讨论中，尽管其他单克隆抗体已被使用，结果相似。我们描述了一系列9例患者，包括2例成人起病RE。利妥昔单抗在病程早期使用（范围1-108个月，平均32个月）。在9例RE患者中，利妥昔单抗对他们的癫痫发作负担有显著的益处。3例患者癫痫发作自由。4/9出现持续部分性癫痫（EPC）， 4/9未发现局灶性运动缺陷。9月2日未出现神经功能缺损进展，5月9日神经影像学进展的证据被利妥昔单抗终止，支持在RE患者中早期使用利妥昔单抗。
Rituximab in the early course of the disease results in good control of seizures and EPC. Rituximab can reverse or stabilize motor deficits as well as MRI changes and halt progression of disease. Rituximab is a safe, efficacious, well tolerated and rapidly acting drug in RE, and can be used as a steroid sparing agent. Rasmussen's encephalitis (RE) is a rare chronic inflammatory disease of the brain resulting in unilateral hemispheric atrophy with drug-resistant focal epilepsy associated with a variable degree of progressive hemiparesis and cognitive decline. The precise etiology of RE is unknown but presumed to have a neuroinflammatory pathobiological basis. Only surgery halts progression of the disease, but may occur at the expense of a fixed but otherwise inevitable neurological deficit. Therefore, the question of medical management is an important consideration. Reports of rituximab use in patients with RE were presented at the American Epilepsy Society annual meeting in 2008. Good published evidence for its usage has been very slow to emerge since then. However, rituximab continues to be listed in discussions of treatment options for patients with RE, though other monoclonal antibodies have since been used with comparable outcomes. We describe a series of nine patients including two with adult-onset RE. Rituximab was used early in the disease course (range 1–108 months; mean 32 months). Of nine patients with RE, there was significant benefit in their seizure burden with rituxamab. Seizure freedom occurred in 3 patients. Epilepsia partialis continua (EPC) was present in 4/9 and no focal motor deficit noted in 4/9. No progression of a neurological deficit was present in 2/9 and evidence of progression with neuroimaging was terminated with rituxamab in 5/9 supporting early use of rituxamab in patients with RE.
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影响因子：
3.4
作者：
Castellano JF;Meyer JA;Lado FA
通讯作者：
Lado FA
影响因子：
9.9
作者：
RASMUSSEN, T;OLSZEWSKI, J;LLOYDSMITH, D
通讯作者：
LLOYDSMITH, D
DOI：
10.3390/diagnostics12020502
发表时间：
2022-02-15
期刊：
Diagnostics (Basel, Switzerland)
影响因子：
--
作者：
Doniselli FM;Deleo F;Criscuolo S;Stabile A;Pastori C;Di Giacomo R;Didato G;Chiapparini L;Villani F
通讯作者：
Villani F
影响因子：
9.9
作者：
Granata, T;Gobbi, G;Fusco, L
通讯作者：
Fusco, L
影响因子：
38.1
作者：
Thilo, Barbara;Stingele, Robert;Lang, Nicolas
通讯作者：
Lang, Nicolas