Ofatumumab treatment in new-onset narcolepsy type 1 following SARS-CoV-2 infection

IF 4.4 2区 医学 Q1 CLINICAL NEUROLOGY
Xiaoli Wang, Xinbo Zhang, Na Yuan, Yonghong Liu
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引用次数: 0

Abstract

Objectives

To explore the efficacy of ofatumumab in new onset narcolepsy type 1 following SARS-CoV-2 infection.

Methods

We present a 9-year-old girl who experienced new onset narcolepsy type 1 following SARS-CoV-2 infection. Polysomnography (PSG) followed by a daytime multiple sleep latency test (MSLT) was under taken after admission. A lumbar puncture was performed to evaluate the CSF orexin-A level. We assessed the CSF hypocretin-1 concentration utilizing the RIA kit from Phoenix Pharmaceuticals Inc. HLA typing was performed. Furthermore, we treated the patient with subcutaneous injections of ofatumumab, and followed her for nearly six-month. The CSF orexin-A level, CD19+ and total B cell population were measured before and after treatment.

Results

The girl had experienced SARS-CoV-2 infection 4 months before presentation. After that, she started to experience excessive daytime sleepiness and cataplexy. She also began to experience nightmares and violent behaviors during her nocturnal sleep, which were not present before her SARS-CoV-2 infection. At the same time, she developed obesity and exhibited psychiatric symptoms such as agitation, anxiety, and aggression. MSLT showed a mean sleep latency of 2.7 min, and 5 times sleep onset REM periods. The CSF orexin-A level was pathologically low at 34.06 pg/mL, and she tested positive for HLA-DQB1*06:02. Consequently, a diagnosis of narcolepsy type 1 was confirmed. Before and after treatment with subcutaneous injections of ofatumumab, the CD19+ and total B cell population before treatment and after 1 months showed a significant reduction from 11% and 296 cells per microliter to 0.56% and 11 cells per microliter, respectively. Within a week following ofatumumab therapy, there was a marked improvement in both excessive daytime sleepiness and cataplexy. Notably, her cataplexy was almost entirely resolved following ofatumumab therapy. Her condition remained stable throughout the 9-month follow-up period. She could normally attend school.

Interpretation

The efficacy of ofatumumab in this case provides additional support for an autoimmune etiology in narcolepsy with cataplexy, highlighting the potential involvement of B-cells in its pathophysiology. This understanding will aid in the development of specific immunotherapeutic strategies for early implementation upon disease onset.

Ofatumumab治疗SARS-CoV-2感染后新发1型发作性睡病
目的:探讨ofatumumab治疗SARS-CoV-2感染后新发1型发作性睡病的疗效。方法:我们报告了一名9岁女孩在SARS-CoV-2感染后出现新发1型发作性睡病。入院后进行多导睡眠图(PSG)和日间多次睡眠潜伏期测试(MSLT)。腰椎穿刺评估脑脊液食欲素A水平。我们使用菲尼克斯制药公司的RIA试剂盒评估脑脊液下丘脑分泌素-1浓度。进行HLA分型。此外,我们对患者进行了ofatumumab皮下注射治疗,并对她进行了近6个月的随访。检测治疗前后脑脊液orexin-A水平、CD19+及总B细胞群。结果:该女孩在就诊前4个月已感染SARS-CoV-2。在那之后,她开始经历白天过度嗜睡和昏厥。她还开始在夜间睡眠中经历噩梦和暴力行为,这些在她感染SARS-CoV-2之前是不存在的。与此同时,她出现了肥胖,并表现出躁动、焦虑和攻击性等精神症状。MSLT显示平均睡眠潜伏期为2.7 min,睡眠开始期为REM的5倍。脑脊液促食欲素a病理水平为34.06 pg/mL, HLA-DQB1*06:02阳性。因此,确诊为1型发作性睡病。皮下注射ofatumumab治疗前后,治疗前和1个月后CD19+和总B细胞群分别从11%和296个细胞/微升显著降低到0.56%和11个细胞/微升。在ofatumumab治疗后的一周内,白天过度嗜睡和猝倒都有明显改善。值得注意的是,她的中风在阿图单抗治疗后几乎完全消失。在9个月的随访期间,患者的病情保持稳定。她可以正常上学。解释:本例中ofatumumab的疗效为发作性睡伴猝倒的自身免疫性病因提供了额外的支持,突出了b细胞在其病理生理中的潜在参与。这一认识将有助于开发特异性免疫治疗策略,以便在疾病发作时尽早实施。
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来源期刊
Annals of Clinical and Translational Neurology
Annals of Clinical and Translational Neurology Medicine-Neurology (clinical)
CiteScore
9.10
自引率
1.90%
发文量
218
审稿时长
8 weeks
期刊介绍: Annals of Clinical and Translational Neurology is a peer-reviewed journal for rapid dissemination of high-quality research related to all areas of neurology. The journal publishes original research and scholarly reviews focused on the mechanisms and treatments of diseases of the nervous system; high-impact topics in neurologic education; and other topics of interest to the clinical neuroscience community.
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