Case report: Cilioretinal artery occlusion combined with central retinal vein occlusion as the initial presentation of pulmonary arterial hypertension.

IF 3.1 3区 医学 Q1 MEDICINE, GENERAL & INTERNAL
Frontiers in Medicine Pub Date : 2024-12-20 eCollection Date: 2024-01-01 DOI:10.3389/fmed.2024.1493834
Yane Gao, Xiaofeng Xie, Jiao Li, Qingshuai Mu, Xingrong Wang, Shuya Wang
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Abstract

Background: Pulmonary arterial hypertension (PAH) is characterized by elevated pulmonary artery pressure and vascular resistance, leading to systemic venous hypertension and potential right heart failure. These elevated pressures can extend to ocular veins, resulting in complications such as central retinal vein occlusion (CRVO). This case report highlights a rare instance of CRVO combined with cilioretinal artery occlusion (CilRAO), an uncommon ocular manifestation associated with PAH.

Case presentation: A 13-year-old girl with a history of surgically repaired ventricular septal defect presented with sudden vision loss and a central visual field defect. Investigation confirmed CRVO and an unusual concurrent CilRAO. Although laboratory tests were inconclusive, the echocardiographic examination suggested severe pulmonary arterial hypertension (PAH). The patient received treatment with Bosentan and traditional Chinese medicine, which improved her vision to 20/20, though a paracentral scotoma remained.

Conclusion: This case underscores the critical link between systemic cardiovascular abnormalities and ocular health in PAH, where elevated venous pressure can result in severe and distinctive ocular manifestations. The co-occurrence of CRVO and CilRAO in this patient highlights the susceptibility of cilioretinal arteries to hemodynamic changes, as these arteries lack autoregulatory capacity. Additionally, this case demonstrates the potential for positive outcomes in ocular lesions with targeted PAH therapy. Importantly, it emphasizes the need for vigilance when managing complex cases where conclusions cannot be drawn solely from ocular findings. A multidisciplinary approach and comprehensive diagnostics are essential for identifying underlying causes, ensuring active treatment, and preventing permanent vision loss and serious systemic complications.

病例报告:以纤毛视网膜动脉闭塞合并视网膜中央静脉闭塞为肺动脉高压的首发表现。
背景:肺动脉高压(PAH)以肺动脉压升高和血管阻力升高为特征,导致全身静脉高压和潜在的右心衰。这些升高的压力可扩展到眼静脉,导致并发症,如视网膜中央静脉阻塞(CRVO)。本病例报告强调了一例罕见的CRVO合并睫状体视网膜动脉闭塞(CilRAO),这是一种罕见的与PAH相关的眼部表现。病例介绍:一名13岁的女孩,有手术修复室间隔缺损的历史,表现为突然视力丧失和中央视野缺损。调查证实CRVO和不寻常的并发CilRAO。虽然实验室检查不确定,超声心动图检查提示严重肺动脉高压(PAH)。患者接受波生坦和中药治疗,视力恢复到20/20,但仍有中心旁暗斑。结论:本病例强调了PAH患者全身心血管异常与眼部健康之间的关键联系,静脉压升高可导致严重而独特的眼部表现。该患者同时出现CRVO和CilRAO,表明纤毛视网膜动脉对血流动力学变化的易感性,因为这些动脉缺乏自我调节能力。此外,该病例表明,靶向多环芳烃治疗眼部病变的潜在积极结果。重要的是,它强调了在处理复杂病例时需要保持警惕,因为这些病例不能仅从观察结果中得出结论。多学科方法和综合诊断对于确定根本原因、确保积极治疗、预防永久性视力丧失和严重的全身并发症至关重要。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Frontiers in Medicine
Frontiers in Medicine Medicine-General Medicine
CiteScore
5.10
自引率
5.10%
发文量
3710
审稿时长
12 weeks
期刊介绍: Frontiers in Medicine publishes rigorously peer-reviewed research linking basic research to clinical practice and patient care, as well as translating scientific advances into new therapies and diagnostic tools. Led by an outstanding Editorial Board of international experts, this multidisciplinary open-access journal is at the forefront of disseminating and communicating scientific knowledge and impactful discoveries to researchers, academics, clinicians and the public worldwide. In addition to papers that provide a link between basic research and clinical practice, a particular emphasis is given to studies that are directly relevant to patient care. In this spirit, the journal publishes the latest research results and medical knowledge that facilitate the translation of scientific advances into new therapies or diagnostic tools. The full listing of the Specialty Sections represented by Frontiers in Medicine is as listed below. As well as the established medical disciplines, Frontiers in Medicine is launching new sections that together will facilitate - the use of patient-reported outcomes under real world conditions - the exploitation of big data and the use of novel information and communication tools in the assessment of new medicines - the scientific bases for guidelines and decisions from regulatory authorities - access to medicinal products and medical devices worldwide - addressing the grand health challenges around the world
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