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儿童 Takayasu 动脉炎以失明为首发表现。

Amaurosis as an initial presentation of Takayasu arteritis in children.

机构信息

Department of Clinical Immunology, Instituto Nacional de Pediatría, Av. Insurgentes Sur 3700-C, Insurgentes Cuicuilco, Coyoacán, México City, 04530, México.

Department of Radiology, Instituto Nacional de Pediatría, México City, México.

出版信息

Rheumatol Int. 2023 Mar;43(3):575-587. doi: 10.1007/s00296-022-05115-2. Epub 2022 Apr 2.

DOI:10.1007/s00296-022-05115-2
PMID:35366089
Abstract

Visual disturbances in Takayasu arteritis (TA) are common but tend to be late manifestations of the disease. However, its presence at diagnosis must alert TA to avoid sight disabilities. Herein, we present two children with TA that debuted with vision loss, and the results of the literature review displayed 58 subjects with vision loss before the diagnosis of TA. The world English literature was reviewed by searching the PubMed database of the National Library of Medicine for the terms "Takayasu Arteritis" and "Blindness" or "Amaurosis fugax", from 190 to 2021. Cases eligible must present vision loss before or at TA diagnosis. Our two patients who presented with amaurosis fulfilled the criteria for TA diagnosis. The first patient had a bilateral and transient visual loss, whereas the second had monocular and permanent amaurosis. Both patients were cursed with hypertension and demonstrated large vessel compromise; their clinical picture improved with corticosteroids and immunosuppressant therapy. We identified in the literature review sixteen patients with TA in case reports and 42 in case series, plus our two cases presented herein with monocular or bilateral vision loss at the time of diagnosis. Previous literature indicated that amaurosis represents a severely advanced disease. Herein, we reported two children with amaurosis as their pivotal symptom; they had significant head and neck vascular alterations, so prompt and aggressive treatment is needed to prevent disease progression and disability. Transient or permanent vision loss must alert the physician to include Takayasu arteritis in the differential diagnosis.

摘要

在多发性大动脉炎(TA)中,视觉障碍较为常见,但往往是疾病的晚期表现。然而,在诊断时出现这种情况必须警惕 TA,以避免视力残疾。在此,我们介绍两例以视力丧失为首发表现的 TA 患儿,同时对文献复习结果显示 58 例在 TA 诊断前出现视力丧失的病例。通过在国家医学图书馆的 PubMed 数据库中检索“Takayasu Arteritis”和“Blindness”或“Amaurosis fugax”,对世界英文文献进行了回顾,检索范围为 190 年至 2021 年。入选病例必须在 TA 诊断前或同时出现视力丧失。我们介绍的两名出现视力丧失的患者均符合 TA 诊断标准。第一个患者出现双侧和短暂的视力丧失,而第二个患者则出现单眼和永久性的失明。这两个患者都患有高血压,并表现出大血管损伤;他们的临床症状在接受皮质类固醇和免疫抑制剂治疗后得到改善。我们在文献复习中发现,16 例 TA 患者为病例报告,42 例为病例系列,加上我们在此介绍的两个在诊断时出现单眼或双眼视力丧失的病例。先前的文献表明,失明代表疾病处于严重晚期。在此,我们报告了两例以失明为首发症状的儿童病例;他们有显著的头颈部血管改变,因此需要及时和积极的治疗,以防止疾病进展和残疾。短暂或永久性的视力丧失必须提醒医生将 TA 纳入鉴别诊断。

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1
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2
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Pulseless and blindness - An ophthalmologist's role in diagnosing Takayasu arteritis: Case series and brief review of literature.无脉症和失明——眼科医生在诊断 Takayasu 动脉炎中的作用:病例系列及文献复习。
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本文引用的文献

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Pulseless and blindness - An ophthalmologist's role in diagnosing Takayasu arteritis: Case series and brief review of literature.无脉症和失明——眼科医生在诊断 Takayasu 动脉炎中的作用:病例系列及文献复习。
Eur J Ophthalmol. 2021 Nov;31(6):3525-3531. doi: 10.1177/1120672121990577. Epub 2021 Feb 12.
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Pediatric vasculitis.小儿血管炎
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