Spontaneous Resolution of a Combined Hamartoma in a One-Year-Old Child.

IF 0.9 4区 医学 Q4 OPHTHALMOLOGY
Marissa Firlie, Shannon Scarboro, C Armitage Harper
{"title":"Spontaneous Resolution of a Combined Hamartoma in a One-Year-Old Child.","authors":"Marissa Firlie, Shannon Scarboro, C Armitage Harper","doi":"10.3928/23258160-20240412-01","DOIUrl":null,"url":null,"abstract":"<p><p>The combined hamartoma of the retina and the retinal pigment epithelium (CHR-RPE) is a rare but histologically benign tumor of which no clear consensus exists regarding its appropriate management. Most commonly CHR-RPE presents unilaterally in children as an elevated, pigmented lesion associated with vascular tortuosity and an epiretinal membrane (ERM). Patients may have decreased visual acuity (VA) in the affected eye, notably if the overlying ERM extends into the macula. For this reason, surgical intervention in the form of ERM removal has been pursued with notable success. Still, those who pursue observation cite the interwoven nature of the overlying glial membrane within the dysplastic retina, complicating surgery and accounting for variability in post-operative VA success. Given the rarity of the tumor coupled with the scarcity of data on its natural evolution, clinicians who pursue observation cannot know for sure whether withholding surgical management is predisposing their patients to worse visual outcomes as compared to those patients who undergo ERM peel as first-line therapy. This case report will discuss an infant with clinically diagnosed CHR-RPE whose lesion showed significant regression after 6 months of observation. This success story should cause ophthalmologists to reconsider the management options at their disposal when faced with a case of CHR-RPE, and to call for further research to better define the risks of observation over surgical intervention. <b>[<i>Ophthalmic Surg Lasers Imaging Retina</i> 2024;55:471-473.]</b>.</p>","PeriodicalId":19679,"journal":{"name":"Ophthalmic surgery, lasers & imaging retina","volume":" ","pages":"471-473"},"PeriodicalIF":0.9000,"publicationDate":"2024-08-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Ophthalmic surgery, lasers & imaging retina","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.3928/23258160-20240412-01","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/5/1 0:00:00","PubModel":"Epub","JCR":"Q4","JCRName":"OPHTHALMOLOGY","Score":null,"Total":0}
引用次数: 0

Abstract

The combined hamartoma of the retina and the retinal pigment epithelium (CHR-RPE) is a rare but histologically benign tumor of which no clear consensus exists regarding its appropriate management. Most commonly CHR-RPE presents unilaterally in children as an elevated, pigmented lesion associated with vascular tortuosity and an epiretinal membrane (ERM). Patients may have decreased visual acuity (VA) in the affected eye, notably if the overlying ERM extends into the macula. For this reason, surgical intervention in the form of ERM removal has been pursued with notable success. Still, those who pursue observation cite the interwoven nature of the overlying glial membrane within the dysplastic retina, complicating surgery and accounting for variability in post-operative VA success. Given the rarity of the tumor coupled with the scarcity of data on its natural evolution, clinicians who pursue observation cannot know for sure whether withholding surgical management is predisposing their patients to worse visual outcomes as compared to those patients who undergo ERM peel as first-line therapy. This case report will discuss an infant with clinically diagnosed CHR-RPE whose lesion showed significant regression after 6 months of observation. This success story should cause ophthalmologists to reconsider the management options at their disposal when faced with a case of CHR-RPE, and to call for further research to better define the risks of observation over surgical intervention. [Ophthalmic Surg Lasers Imaging Retina 2024;55:471-473.].

一名一岁儿童的组合性 Hamartoma 病例的自然消退
视网膜和视网膜色素上皮联合仓鼠神经瘤(CHR-RPE)是一种罕见的组织学良性肿瘤,目前对其适当的治疗方法还没有明确的共识。CHR-RPE 最常见于单侧患儿,表现为隆起的色素性病变,伴有血管迂曲和视网膜上膜(ERM)。患者患眼的视力(VA)可能会下降,尤其是当覆盖的 ERM 扩展到黄斑时。因此,以切除 ERM 为形式的手术干预取得了显著的成功。不过,进行观察的人认为,上覆的胶质膜在发育不良的视网膜内交织在一起,使手术复杂化,也是术后视力成功率存在差异的原因。鉴于该肿瘤的罕见性及其自然演化数据的稀缺性,临床医生无法确定,与接受ERM剥离作为一线治疗的患者相比,拒绝手术治疗是否会导致患者的视力下降。本病例报告将讨论一名临床诊断为 CHR-RPE 的婴儿,经过 6 个月的观察,其病变明显消退。这一成功案例应促使眼科医生在面对 CHR-RPE 病例时重新考虑他们所能选择的治疗方案,并呼吁开展进一步的研究,以更好地界定观察与手术干预相比的风险。[眼科手术激光成像视网膜 2024;55:XX-XX]。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
CiteScore
1.80
自引率
0.00%
发文量
89
期刊介绍: OSLI Retina focuses exclusively on retinal diseases, surgery and pharmacotherapy. OSLI Retina will offer an expedited submission to publication effort of peer-reviewed clinical science and case report articles. The front of the journal offers practical clinical and practice management features and columns specific to retina specialists. In sum, readers will find important peer-reviewed retina articles and the latest findings in techniques and science, as well as informative business and practice management features in one journal.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信