First reported pediatric case of left internal carotid artery stenosis in myelin oligodendrocyte glycoprotein antibody-associated disease

Eri Hasegawa , Jun Kubota , Taku Gomi , Shuntaro Terayama , Taiki Homma , Haruna Suzuki , Yoichi Takemasa , Ryota Saito , Kenta Horimukai , Noriko Takahata
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Abstract

Background

Myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease (MOGAD) mimics the clinical and imaging findings of small-vessel central nervous system (CNS) angiitis. An adult case of MOGAD causing right middle cerebral artery stenosis was reported in 2023. Here, we present the first reported pediatric case of left internal carotid artery stenosis in a patient with MOGAD.

Case presentation

A previously healthy 13-year-old boy presented with a two-day history of fever and headache. He experienced sudden focal-onset impaired awareness tonic seizures on the right side, with right ocular deviation. Seizure activity ceased within 5 min, but unconsciousness and paralysis of the right face and right upper extremity persisted on admission. There were no other abnormal neurological findings. Blood tests revealed mildly elevated levels of inflammatory markers. Cerebrospinal fluid examination revealed a normal protein level of 39.3 mg/dL but an elevated cell count of 154/µL and an oligoclonal band. Fluid-attenuated inversion recovery MRI sequences revealed hyperintensities in the left basal ganglia and left frontoparietal cortex. Magnetic resonance angiography revealed left internal carotid artery stenosis. Subsequently, MOGAD was diagnosed based on a positive MOG antibody test result. He received three courses of methylprednisolone pulse therapy followed by oral prednisolone for 10 weeks. His symptoms, parenchymal brain lesions, and vascular stenosis all improved with treatment.

Discussion/Conclusion

MOGAD may be associated with vascular stenosis by inducing a perivascular immune response. MOGAD may mimic CNS angiitis, including that of medium- and large-sized vessels. The presence of vascular stenosis does not rule out MOGAD.

首例报道的髓鞘少突胶质细胞糖蛋白抗体相关疾病左侧颈内动脉狭窄儿科病例
背景髓鞘少突胶质细胞糖蛋白(MOG)抗体相关疾病(MOGAD)模仿小血管中枢神经系统(CNS)血管炎的临床和影像学表现。2023 年报道了一例 MOGAD 导致右侧大脑中动脉狭窄的成人病例。在此,我们介绍首例MOGAD患者左侧颈内动脉狭窄的儿科病例。病例介绍一名之前身体健康的13岁男孩,两天前出现发热和头痛。他突然出现右侧局灶性意识障碍强直发作,伴右眼偏斜。发作活动在 5 分钟内停止,但入院时仍昏迷不醒,右脸和右上肢瘫痪。神经系统没有其他异常发现。血液检查显示炎症标志物水平轻度升高。脑脊液检查显示蛋白质水平正常,为 39.3 mg/dL,但细胞计数升高至 154 个/μL,并出现一条寡克隆带。体液减弱反转恢复磁共振成像序列显示左侧基底节和左侧额顶叶皮层高密度。磁共振血管造影显示左侧颈内动脉狭窄。随后,根据 MOG 抗体检测阳性结果,他被诊断为 MOGAD。他接受了三个疗程的甲基强的松龙脉冲治疗,随后口服强的松龙10周。讨论/结论MOGAD可能通过诱导血管周围免疫反应而与血管狭窄有关。MOGAD 可模拟中枢神经系统血管炎,包括中型和大型血管炎。血管狭窄的存在并不能排除 MOGAD 的可能性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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