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神经生长因子治疗黏蛋白性类天疱疮导致神经营养性角膜病变和角膜缘干细胞缺乏患者后角膜浅层斑块形成

Corneal Superficial Plaque Formation After Recombinant Human Nerve Growth Factor Use in a Patient With Neurotrophic Keratopathy and Limbal Stem Cell Deficiency From Mucous Membrane Pemphigoid.

机构信息

Department of Ophthalmology, Massachusetts Eye and Ear Institute, Harvard Medical School, Boston, MA.

出版信息

Cornea. 2024 Jul 1;43(7):899-902. doi: 10.1097/ICO.0000000000003442. Epub 2023 Nov 27.

Abstract

PURPOSE

The aim of this study was to report a rare observation of corneal superficial plaque formation after topical recombinant human nerve growth factor (rhNGF) treatment for a nonhealing epithelial defect in a patient with advanced mucous membrane pemphigoid, limbal stem cell deficiency, and neurotrophic keratopathy.

METHODS

This study was a case report.

RESULTS

A 72-year-old man with a complex course of mucous membrane pemphigoid, leading to cicatrizing keratoconjunctivits, limbal stem cell deficiency, and neurotrophic keratopathy presented with a recurrent persistent epithelial defect in the right eye. After a long course of unsuccessful epithelial healing, despite various treatment modalities, he was administered topical rhNGF (cenegermin 0.002%; Oxervate, Dompé US Inc., Boston, MA) which successfully resolved the epithelial defect. However, on day 22 posttreatment, an unusual white, thick, adherent corneal superficial plaque formed. rhNGF was stopped and the plaque was carefully removed. Subsequently, there was no recurrence, and the patient's epithelial healing remained stable.

CONCLUSIONS

Although the successful resolution of the persistent epithelial defect with rhNGF administration was notable, the development of the unusual epithelial overgrowth emphasizes the importance of vigilant monitoring and evaluation when using rhNGF in complex ocular conditions. Making informed decisions on the timing of discontinuing rhNGF can lead to desirable effects of the drug while mitigating additional side effects when managing such challenging cases.

摘要

目的

本研究旨在报告一例罕见病例,在一名患有进展性黏膜类天疱疮、边缘干细胞缺乏症和神经营养性角膜病变的患者中,使用局部重组人神经生长因子(rhNGF)治疗不愈合的上皮缺损后,出现角膜浅层斑块形成。

方法

本研究为病例报告。

结果

一名 72 岁男性,患有复杂的黏膜类天疱疮,导致瘢痕性角结膜炎、边缘干细胞缺乏症和神经营养性角膜病变,右眼出现反复持续性上皮缺损。尽管采用了各种治疗方法,但上皮愈合仍未成功,经过长时间的治疗后,他开始接受局部 rhNGF(cenegermin 0.002%;Oxervate,Dompé US Inc.,Boston,MA)治疗,成功解决了上皮缺损问题。然而,在治疗后第 22 天,出现了一种不寻常的白色、厚而黏附的角膜浅层斑块。rhNGF 被停用,斑块被小心地清除。随后,没有再次出现斑块,患者的上皮愈合保持稳定。

结论

尽管 rhNGF 治疗成功解决了持续性上皮缺损问题,但异常上皮过度生长的发生强调了在复杂眼部疾病中使用 rhNGF 时进行警惕监测和评估的重要性。在管理此类具有挑战性的病例时,及时决定停用 rhNGF 可以发挥药物的理想效果,同时减轻其他不良反应。

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