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早发型获得性脱髓鞘综合征(ADS)会掩盖小儿白塞病吗?一例病例报告。

Can early-onset acquired demyelinating syndrome (ADS) hide pediatric Behcet's disease? A case report.

作者信息

Pozzato Mattia, Dilena Robertino, Rogani Greta, Beretta Gisella, Torreggiani Sofia, Lanni Stefano, Tozzo Alessandra, Andreetta Francesca, Cavalcante Paola, Triulzi Fabio, Martinelli Boneschi Filippo, Minoia Francesca, Filocamo Giovanni

机构信息

Neurology Unit & MS Centre, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

Dino Ferrari Centre, Neuroscience Section, Department of Pathophysiology and Transplantation, University of Milan, Milan, Italy.

出版信息

Front Pediatr. 2023 Jun 23;11:1175584. doi: 10.3389/fped.2023.1175584. eCollection 2023.

Abstract

Behcet's disease (BD) is a rare vasculitis characterized by multisystemic inflammation. Central nervous system (CNS) involvement is rare and heterogeneous, particularly in the pediatric population. A diagnosis of neuro-Behcet could be highly challenging, especially if neurological manifestations precede other systemic features; however, its timely definition is crucial to prevent long-term sequelae. In this study, we describe the case of a girl who, at 13 months of age, presented with a first episode of encephalopathy compatible with acute disseminated encephalomyelitis, followed, after 6 months, by a neurological relapse characterized by ophthalmoparesis and gait ataxia, in association with new inflammatory lesions in the brain and spinal cord, suggesting a neuromyelitis optica spectrum disorder. The neurological manifestations were successfully treated with high-dose steroids and intravenous immunoglobulins. In the following months, the patient developed a multisystemic involvement suggestive of Behcet's disease, characterized by polyarthritis and uveitis, associated with HLA-B51 positivity. The challenge presented by this unique case required a multidisciplinary approach involving pediatric neurologists, neuro-radiologists, and pediatric rheumatologists, with all of these specialists creating awareness about early-onset acquired demyelinating syndromes (ADSs). Given the rarity of this presentation, we performed a review of the literature focusing on neurological manifestations in BD and differential diagnosis of patients with early-onset ADS.

摘要

白塞病(BD)是一种罕见的血管炎,其特征为多系统炎症。中枢神经系统(CNS)受累罕见且具有异质性,在儿童群体中尤为如此。神经白塞病的诊断可能极具挑战性,特别是当神经症状先于其他全身症状出现时;然而,及时明确诊断对于预防长期后遗症至关重要。在本研究中,我们描述了一名13个月大女孩的病例,她首次出现与急性播散性脑脊髓炎相符的脑病发作,6个月后出现以眼球运动麻痹和步态共济失调为特征的神经复发,并伴有脑和脊髓新的炎症性病变,提示视神经脊髓炎谱系障碍。神经症状通过大剂量类固醇和静脉注射免疫球蛋白成功得到治疗。在接下来的几个月里,患者出现了提示白塞病的多系统受累,表现为多关节炎和葡萄膜炎,并伴有HLA - B51阳性。这个独特病例带来的挑战需要儿科神经科医生、神经放射科医生和儿科风湿病学家采取多学科方法,所有这些专家都提高了对早发性获得性脱髓鞘综合征(ADSs)的认识。鉴于这种表现的罕见性,我们对文献进行了综述,重点关注白塞病的神经症状以及早发性ADS患者的鉴别诊断。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac93/10327559/645bfec20714/fped-11-1175584-g001.jpg

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