Ambia Ayesha R, AlZahrani Norah, Almakadma Abdul Hakim, Elgazzar Tasnim A, Almustanyir Sami
College of Medicine, Alfaisal University, Riyadh, SAU.
Neurology, Prince Mohammed Bin Abdulaziz Hospital, RIyadh, SAU.
Cureus. 2022 Jan 18;14(1):e21382. doi: 10.7759/cureus.21382. eCollection 2022 Jan.
Chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids (CLIPPERS) is a rare disease with an unknown etiology which most commonly results in subacute diplopia and ataxia. Diagnosis is achieved through a triad of the following findings: lymphocytic pleocytosis with increased CD4+ T cells on cerebrospinal fluid (CSF) analysis; perivascular punctate and curvilinear hemorrhages in the pons, medulla, or cerebellum on magnetic resonance imaging (MRI) with contrast; and the cessation of symptoms after the initiation of corticosteroids. Here, we report the case of a 23-year-old male who presented with non-specific signs and symptoms, including diffuse weakness in all limbs, ataxia, and slurred speech. The diagnosis was achieved through a contrast MRI of the brain, suggestive of brainstem encephalitis, and a CSF analysis, which revealed elevated glucose and protein levels. Intravenous methylprednisolone was administered for five days and resulted in acute improvement of the patient's clinical status. Repeat CSF analysis and MRI of the brain with contrast two weeks later showed resolution of previous findings. CLIPPERS syndrome is a newly identified disease thought to cause a predominantly inflammatory reaction in the pons, medulla, cerebellum, and supratentorial region. MRI with contrast tends to reveal a "salt and pepper appearance" in a punctate and curvilinear fashion. The hallmark of treatment is corticosteroid therapy, and discontinuation of therapy should be done with caution as relapse of the syndrome with corticosteroid withdrawal has been documented.
类固醇反应性桥脑周围血管强化慢性淋巴细胞性炎症(CLIPPERS)是一种病因不明的罕见疾病,最常见的表现为亚急性复视和共济失调。诊断通过以下三项发现来实现:脑脊液(CSF)分析显示淋巴细胞增多伴CD4 + T细胞增加;磁共振成像(MRI)增强扫描显示脑桥、延髓或小脑出现血管周围点状和曲线状出血;使用皮质类固醇后症状缓解。在此,我们报告一例23岁男性患者,其表现为非特异性体征和症状,包括四肢弥漫性无力、共济失调和言语含糊。通过脑部增强MRI提示脑干脑炎以及CSF分析显示葡萄糖和蛋白质水平升高得以确诊。静脉注射甲基强的松龙5天,患者临床状态急性改善。两周后重复进行CSF分析和脑部增强MRI显示先前的表现消失。CLIPPERS综合征是一种新发现的疾病,被认为主要在脑桥、延髓、小脑和幕上区域引起炎症反应。增强MRI往往显示出点状和曲线状的“椒盐样外观”。治疗的标志是皮质类固醇疗法,由于已有文献记载停用皮质类固醇会导致该综合征复发,因此停药应谨慎。