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一名16岁男性患者,以急性后部多灶性扁平状色素上皮病变为表现的脑动脉炎

Cerebral vasculitis presenting as acute posterior multifocal placoid pigment epitheliopathy in a 16-year-old male.

作者信息

Ayala Rodríguez Sofía C, Ramirez Marquez Estefania, Torres-Rosa Ángel G, Ramirez Marquez Jerome A, Boada Roberto, Oliver Armando L, Rodríguez-Rosa Ricardo E

机构信息

University of Puerto Rico Medical Sciences Campus, Department of Ophthalmology, Paseo Dr. Jose Celso Barbosa, San Juan, PR, USA.

University of South Alabama, College of Medicine, Department of Radiology, Mobile, AL, USA.

出版信息

Am J Ophthalmol Case Rep. 2024 Jul 20;36:102106. doi: 10.1016/j.ajoc.2024.102106. eCollection 2024 Dec.

DOI:10.1016/j.ajoc.2024.102106
PMID:39161377
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11331703/
Abstract

PURPOSE

To report on a case of the successful treatment of Acute Posterior Multifocal Placoid Pigment Epitheliopathy (APMPPE) in a pediatric patient with a prior diagnosis of cerebral vasculitis.

OBSERVATIONS

A 16-year-old male with a prior diagnosis of cerebral vasculitis presented without ocular complaints. Visual acuity was 20/20, and color vision remained normal. Fundus examination revealed yellowish-white placoid lesions and retinal pigmented epithelial changes involving the posterior pole. A work-up including a rapid plasma reagin test, complete cell blood count, comprehensive metabolic panel, and urinalysis was within normal limits. A head computed tomography angiography without contrast and a brain magnetic resonance imaging scan were compatible with acute and past episodes of ischemia. Ancillary testing was compatible with an assessment APMPPE. Immunosuppressive and monoclonal antibody therapy resulted in the improvement and remission without residual neurologic deficits and with a BCVA of 20/20.

CONCLUSIONAND IMPORTANCE

This case suggests that a diagnosis of cerebral vasculitis should prompt physicians to consider an ophthalmic evaluation that includes a dilated fundus exam, regardless of the presence or absence of ocular symptoms. Ophthalmic findings may affect the diagnostic processes, particularly concerning infectious and non-infectious etiologies, or potentially neoplastic diseases.

摘要

目的

报告一例先前诊断为脑血管炎的儿科患者成功治疗急性后极部多灶性鳞状色素上皮病变(APMPPE)的病例。

观察结果

一名先前诊断为脑血管炎的16岁男性患者,无眼部不适症状。视力为20/20,色觉正常。眼底检查发现后极部有黄白色鳞状病变及视网膜色素上皮改变。包括快速血浆反应素试验、全血细胞计数、综合代谢指标和尿液分析在内的检查均在正常范围内。头颅计算机断层血管造影(无造影剂)和脑部磁共振成像扫描结果与急性和既往缺血发作相符。辅助检查结果与APMPPE的评估相符。免疫抑制和单克隆抗体治疗使病情得到改善并缓解,无残留神经功能缺损,最佳矫正视力为20/20。

结论与意义

该病例表明,无论有无眼部症状,脑血管炎的诊断都应促使医生考虑进行包括散瞳眼底检查在内的眼科评估。眼科检查结果可能会影响诊断过程,尤其是在涉及感染性和非感染性病因或潜在肿瘤性疾病方面。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/00f23d0b1399/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/3dd7538f6fa7/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/de44971539df/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/00f23d0b1399/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/3dd7538f6fa7/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/de44971539df/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be57/11331703/00f23d0b1399/gr3.jpg

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The HAVEN study-hydroxychloroquine in ANCA vasculitis evaluation-a multicentre, randomised, double-blind, placebo-controlled trial: study protocol and statistical analysis plan.HAVEN 研究-羟氯喹在 ANCA 血管炎评估中的作用:一项多中心、随机、双盲、安慰剂对照试验:研究方案和统计分析计划。
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Acute Posterior Multifocal Placoid Pigment Epitheliopathy Complicated by Fatal Cerebral Vasculitis.急性后极部多发性斑状色素上皮病变合并致命性脑血管炎。
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Untreated Acute Posterior Multifocal Placoid Pigment Epitheliopathy (APMPPE): a case series.
未经治疗的急性后极部多灶性鳞状色素上皮病变(APMPPE):病例系列
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Acute Posterior Multifocal Placoid Pigment Epitheliopathy Associated with Stroke: A Case Report and Review of the Literature.与中风相关的急性后极部多灶性鳞状色素上皮病变:一例报告及文献复习
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Delayed recurrent stroke in a young patient with acute posterior multifocal placoid pigment epitheliopathy.年轻患者急性后极部多发性斑状色素上皮病变后迟发性复发性中风。
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