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Review
. 2023 May 1;42(5):645-647.
doi: 10.1097/ICO.0000000000003211. Epub 2022 Dec 19.

Recurrent Lisch Epithelial Corneal Dystrophy Treated With 5-Fluorouracil: A Case Report and Review of the Literature

Mona M Amer  1 , Karen Arze  1 , Anat Galor  1   2 , Yoseph Sayegh  1 , Sander S Dubovy  1 , Carol L Karp  1
Affiliations
Review

Recurrent Lisch Epithelial Corneal Dystrophy Treated With 5-Fluorouracil: A Case Report and Review of the Literature

Mona M Amer et al. Cornea. .

Abstract

Purpose: The aim of the study was to describe a case of Lisch epithelial corneal dystrophy (LECD), review its clinical and histopathological features and diagnostic imaging, and introduce a novel treatment approach using topical 5-fluorouracil (5-FU).

Methods: A 65-year-old woman presented with a recurrent left-sided corneal lesion consistent with LECD. The lesion was evaluated clinically, with high-resolution optical coherence tomography (HR-OCT), and histologically. The lesion was successfully treated with two 1-week cycles of topical 5-FU.

Results: Slit-lamp examination showed an opalescent, whorl-shaped corneal lesion. HR-OCT revealed a trapezoidal area of normal thickness epithelial hyperreflectivity. Histopathology demonstrated a mucosal epithelium with foamy cytoplasm and increased cell size consistent with LECD. Treatment with topical 5-FU resulted in marked clearance of the corneal lesion on slit-lamp examination and HR-OCT.

Conclusions: 5-FU may be considered as a treatment option for LECD.

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Conflict of interest statement

Drs. Karp and Galor have a pending PCT/US2022/029842. All other authors have no other funding or conflict of interest to disclose.

Figures

Figure 1:
Figure 1:
Histopathological analysis of corneal lesion A. Enlarged corneal epithelial cells with foam-filled cytoplasm (arrow) are present adjacent to the well-demarcated, unremarkable epithelium (asterisk). (Hematoxylin-eosin stain, original magnification x400). B. Granules within the cytoplasm are positive with PAS stain (arrow). (Periodic acid-Schiff stain, original magnification x400).
Figure 2:
Figure 2:
Slit lamp images before and after treatment with 5-FU A. Feathery, whorled paracentral corneal epithelial opacification (white arrow). Inset shows clustered, intraepithelial microcysts on retroillumination. B. Clinical resolution of LECD lesion after 2 cycles (ie, 2 months) of 5-FU. Each cycle consisted of one week of treatment with four times daily application, followed by three weeks off.
Figure 3:
Figure 3:
Anterior segment HR-OCT images before and after treatment with 5-FU. A. Non-thickened, trapezoidal shaped, hyperreflective epithelium (white arrow) of corneal lesion before 5-FU treatment initiation. Inset shows location of raster scan. B. Normalized thin dark epithelium (white arrow) of corneal lesion after treatment with 2 cycles (ie, 2 months) of 5-FU. Each cycle consisted of one week of treatment with four times daily application, followed by three weeks off. Inset shows location of raster scan.
See this image and copyright information in PMC

References

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