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眼阵挛-肌阵挛-共济失调综合征:常规疫苗接种后的罕见后果。

Opsoclonus-Myoclonus-Ataxia Syndrome: A Rare Outcome Following Routine Vaccinations.

作者信息

Leuzinger-Dias Catarina, Ferrão Tomás, Rebimbas Sandra, Palavra Filipe, Amaral Joana

机构信息

Neuropediatrics, Centre for Child Development, Hospital Pediátrico, Unidade Local de Saúde de Coimbra, Coimbra, PRT.

Pediatrics, Unidade Local de Saúde da Região de Aveiro, Aveiro, PRT.

出版信息

Cureus. 2024 Nov 25;16(11):e74413. doi: 10.7759/cureus.74413. eCollection 2024 Nov.

DOI:10.7759/cureus.74413
PMID:39723301
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11669349/
Abstract

Opsoclonus-myoclonus-ataxia syndrome (OMAS) is a rare neurological disorder, affecting approximately 0.18 per million individuals annually. It presents with a triad of opsoclonus, myoclonus, and ataxia, often including cognitive dysfunction and behavioral disturbances. Even though OMAS is non-fatal, it frequently follows a chronic-relapsing course, resulting in long-term neuropsychological sequelae in over 50% of patients. Diagnosis is challenging due to variable symptom presentation and the lack of specific diagnostic markers. We report a case of a two-month-old infant who developed OMAS after routine vaccinations, a rare post-vaccination occurrence. Initial treatment with high-dose corticosteroids successfully resolved symptoms, but the patient experienced a relapse during medication tapering. The relapse occurred following another vaccination, suggesting a potential link between the immune response triggered by vaccination and OMAS symptoms. Despite the early relapse, the patient had a favorable recovery, with normal neurodevelopment at 24 months and no further relapses. This case raises awareness of the rare autoimmune post-vaccine reaction as a possible etiology of OMAS, which is infrequently reported in the literature. With this report, we aimed to underscore the importance of early recognition of OMAS, particularly in infants, and highlight the potential for a positive outcome with timely diagnosis and treatment.

摘要

眼阵挛-肌阵挛-共济失调综合征(OMAS)是一种罕见的神经系统疾病,每年影响约百万分之0.18的个体。它表现为眼阵挛、肌阵挛和共济失调三联征,常伴有认知功能障碍和行为紊乱。尽管OMAS并非致命性疾病,但它通常呈慢性复发病程,超过50%的患者会出现长期神经心理后遗症。由于症状表现多样且缺乏特异性诊断标志物,诊断具有挑战性。我们报告一例两个月大的婴儿在常规疫苗接种后发生OMAS的病例,这是一种罕见的疫苗接种后情况。初始使用高剂量皮质类固醇治疗成功缓解了症状,但患者在减药过程中复发。复发发生在另一次疫苗接种之后,提示疫苗引发的免疫反应与OMAS症状之间可能存在联系。尽管早期复发,但患者恢复良好,24个月时神经发育正常且未再复发。该病例提高了人们对罕见的自身免疫性疫苗接种后反应作为OMAS可能病因的认识,这在文献中鲜有报道。通过本报告,我们旨在强调早期识别OMAS的重要性,尤其是在婴儿中,并突出及时诊断和治疗带来良好预后的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/3038ded29dc0/cureus-0016-00000074413-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/a81dec9ea304/cureus-0016-00000074413-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/a68972f6eaff/cureus-0016-00000074413-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/1f7c09d14996/cureus-0016-00000074413-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/3038ded29dc0/cureus-0016-00000074413-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/a81dec9ea304/cureus-0016-00000074413-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/a68972f6eaff/cureus-0016-00000074413-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/1f7c09d14996/cureus-0016-00000074413-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e23/11669349/3038ded29dc0/cureus-0016-00000074413-i04.jpg

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本文引用的文献

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Diagnosis and Management of Opsoclonus-Myoclonus-Ataxia Syndrome in Children: An International Perspective.儿童眼阵挛-肌阵挛-共济失调综合征的诊断与管理:国际视角
Neurol Neuroimmunol Neuroinflamm. 2022 Mar 8;9(3). doi: 10.1212/NXI.0000000000001153. Print 2022 May.
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Opsoclonus-Myoclonus Syndrome in Children and Adolescents: A Therapeutic Challenge.儿童和青少年的眼阵挛-肌阵挛综合征:一项治疗挑战。
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