Bilateral Vasoproliferative Tumors in Usher Syndrome.

IF 0.5 Q4 OPHTHALMOLOGY
Case Reports in Ophthalmology Pub Date : 2025-03-25 eCollection Date: 2025-01-01 DOI:10.1159/000542415
Francesco Pichi, Federico Rissotto, Khalid N Qadha, Scott D Smith, Arif O Khan
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引用次数: 0

Abstract

Introduction: Vasoproliferative tumors (VPTs) can be associated with retinal dystrophy, but there are few well-documented cases and even fewer linked to Usher syndrome. Most reported cases are unilateral. The purpose of this report was to describe a case of bilateral sequential VPT in the context of MYO7A-related Usher syndrome.

Case report: A 13-year-old female presented with pain in her left eye and no light perception in that eye. She was found to have left eye advanced neovascular glaucoma related to VPT in the context of MYO7A-related Usher syndrome. Intraocular pressure and pain were controlled with external diode cyclodestruction. Almost 3 years later, early asymptomatic VPT was noted in the right eye during follow-up and was treated with laser photocoagulation.

Conclusion: Although VPT is typically unilateral, affected patients need careful follow-up for possible bilateral sequential disease. Early treatment avoids blinding complications.

Usher综合征的双侧血管增生性肿瘤。
血管增殖性肿瘤(vpt)可能与视网膜营养不良有关,但很少有充分记录的病例,与Usher综合征有关的病例更少。大多数报告的病例为单侧。本报告的目的是描述一例myo7a相关Usher综合征的双侧序贯VPT。病例报告:一名13岁女性,以左眼疼痛及无光知觉为主诉。在myo7a相关Usher综合征的背景下,她被发现患有与VPT相关的左眼晚期新生血管性青光眼。采用外置二极管环破坏术控制眼压和疼痛。近3年后,随访中发现右眼早期无症状VPT,采用激光光凝治疗。结论:虽然VPT是典型的单侧病变,但对于可能的双侧序贯性疾病,患者需要仔细随访。早期治疗可避免致盲并发症。
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来源期刊
CiteScore
0.90
自引率
0.00%
发文量
129
审稿时长
12 weeks
期刊介绍: This peer-reviewed online-only journal publishes original case reports covering the entire spectrum of ophthalmology, including prevention, diagnosis, treatment, toxicities of therapy, supportive care, quality-of-life, and survivorship issues. The submission of negative results is strongly encouraged. The journal will also accept case reports dealing with the use of novel technologies, both in the arena of diagnosis and treatment. Supplementary material is welcomed. The intent of the journal is to provide clinicians and researchers with a tool to disseminate their personal experiences to a wider public as well as to review interesting cases encountered by colleagues all over the world. Universally used terms can be searched across the entire growing collection of case reports, further facilitating the retrieval of specific information. Following the open access principle, the entire contents can be retrieved at no charge, guaranteeing easy access to this valuable source of anecdotal information at all times.
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