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双侧特发性角膜瘢痕患者板层角膜移植术后的长期随访。

Long-term follow-up after lamellar keratoplasty in a patient with bilateral idiopathic corneal keloid.

机构信息

Department of Ophthalmology, Yamaguchi University Graduate School of Medicine, Yamaguchi, Japan.

出版信息

Cornea. 2011 Dec;30(12):1491-4. doi: 10.1097/ICO.0b013e31822018f2.

DOI:10.1097/ICO.0b013e31822018f2
PMID:22067103
Abstract

PURPOSE

To report a case of bilateral idiopathic corneal keloid.

METHODS

Retrospective review of clinical features, histopathological findings, clinical management, and outcome.

RESULTS

A 2-year-old boy with bilateral corneal keloid was treated with lamellar keratoplasty and tranilast eye drops. Peripheral localized white corneal nodules had been present bilaterally since the age of approximately 6 months in the absence of any history of trauma, inflammatory disease, or relevant family history. Pathological examination of the excised corneal buttons revealed myofibroblast proliferation (positive staining for α-smooth muscle actins), a haphazard arrangement of collagen bundles, and the absence of inflammatory cells. On the basis of these findings, a diagnosis of corneal keloid was assigned. The size of the corneal lesion in the right eye decreased in response to therapy with tranilast eye drops. Lamellar keratoplasty resulted in improved bilateral visual acuity, which was maintained at the 12-year follow-up.

CONCLUSIONS

This report describes a very rare case of bilateral corneal keloid in the absence of trauma or inflammation that was diagnosed by histological and immunohistochemical examination and electron microscopy. Good visual acuity was maintained over an extended period of postsurgery follow-up. Tranilast may represent a novel adjuvant therapy for corneal keloid.

摘要

目的

报告一例双侧特发性角膜瘢痕瘤病例。

方法

回顾性分析临床特征、组织病理学发现、临床治疗及预后。

结果

一名 2 岁男孩双侧角膜瘢痕瘤,行板层角膜移植术联合曲尼司特滴眼液治疗。该男孩自约 6 月龄起双侧出现局限性周边白色角膜结节,无外伤、炎症性疾病或相关家族史。切除的角膜纽扣组织病理学检查显示肌纤维母细胞增生(α-平滑肌肌动蛋白染色阳性),胶原束排列杂乱,无炎症细胞。根据这些发现,诊断为角膜瘢痕瘤。右眼的角膜病变大小对曲尼司特滴眼液治疗有反应,其大小减小。板层角膜移植术后双侧视力均得到改善,并在 12 年随访时得到维持。

结论

本报告描述了一例罕见的双侧无创伤和炎症性角膜瘢痕瘤病例,通过组织学、免疫组织化学和电子显微镜检查进行诊断。术后长期随访视力保持良好。曲尼司特可能是一种治疗角膜瘢痕瘤的新辅助治疗方法。

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