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10 岁男孩 Coats 病伴视网膜前黄斑纤维膜自发性剥脱的长期随访:病例报告

Long-term follow-up of a case of Coats disease in a 10-year-old boy with spontaneous peeling of preretinal macular fibrosis: a case report.

机构信息

Department of Ophthalmology and Visual Science, Kochi Medical School, Kochi University, Kohasu, Oko-cho, Nankoku City, Kochi, 783-8505, Japan.

出版信息

BMC Ophthalmol. 2022 Apr 27;22(1):194. doi: 10.1186/s12886-022-02414-x.

DOI:10.1186/s12886-022-02414-x
PMID:35477388
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9044609/
Abstract

BACKGROUND

Coats disease is a retinal vascular disorder characterized by aneurysms and telangiectasias. Macular fibrosis is a complication of Coats disease that results in vision loss. Macular fibrosis rarely develops in the natural course and often occurs after treatment with intravitreal bevacizumab, photocoagulation, or cryotherapy. Here, we have described an unusual case of spontaneous peeling of preretinal macular fibrosis in a patient with untreated Coats disease.

CASE PRESENTATION

A 10-year-old Japanese boy presented with vision loss in his left eye. The patient's left visual acuity was 20/28. Fundus examination of his left eye revealed thick preretinal macular fibrosis around the optic disc and macula. In addition, retinal telangiectasis, microaneurysms, hard exudates, and retinal hemorrhages were observed in the left peripheral temporal retina. We diagnosed his condition as Coats disease with preretinal macular fibrosis. Two months later, optical coherence tomography revealed preretinal macular fibrosis detachment at the foveal lesion without any treatment. During follow-up, preretinal macular fibrosis at the macular lesion was completely detached. Further, posterior vitreous detachment was observed and the shape of the macula and the patient's left visual acuity had improved.

CONCLUSIONS

In our case, both formation and spontaneous peeling of preretinal macular fibrosis occurred without any treatment for Coats disease, which is an unusual finding. Vitreous changes might have occurred during the natural clinical course, causing subsequent posterior vitreous detachment and resulting in spontaneous peeling of fibrosis.

摘要

背景

Coats 病是一种以动脉瘤和毛细血管扩张为特征的视网膜血管疾病。黄斑部纤维化是 Coats 病的一种并发症,可导致视力丧失。黄斑部纤维化在自然病程中很少发生,通常发生在玻璃体内注射贝伐单抗、光凝或冷冻治疗后。在此,我们描述了一例未经治疗的 Coats 病患者自发剥除黄斑前膜纤维化的罕见病例。

病例介绍

一名 10 岁日本男孩因左眼视力下降就诊。患者左眼视力为 20/28。左眼眼底检查发现视盘和黄斑周围有厚的黄斑前膜纤维化。此外,左眼颞侧周边视网膜还观察到视网膜毛细血管扩张、微动脉瘤、硬性渗出和视网膜出血。我们诊断他患有 Coats 病伴黄斑前膜纤维化。两个月后,光学相干断层扫描显示黄斑病变处的黄斑前膜纤维化无任何治疗而脱离。随访过程中,黄斑病变处的黄斑前膜纤维化完全脱离。此外,还观察到后玻璃体脱离,黄斑形状和患者左眼视力均有所改善。

结论

在我们的病例中,Coats 病未经治疗即出现黄斑前膜纤维化的形成和自发剥除,这是一种罕见的表现。玻璃体变化可能在自然病程中发生,导致随后的后玻璃体脱离,从而导致纤维化的自发剥除。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6c3e/9044609/dd8750788a23/12886_2022_2414_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6c3e/9044609/dd8750788a23/12886_2022_2414_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6c3e/9044609/dd8750788a23/12886_2022_2414_Fig1_HTML.jpg

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本文引用的文献

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Am J Ophthalmol Case Rep. 2019 Jul 9;15:100508. doi: 10.1016/j.ajoc.2019.100508. eCollection 2019 Sep.
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Treatment of Coats' disease: an analysis of pooled results.科茨病的治疗:汇总结果分析
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