Sir,

Corneal epithelial dysmaturation is clinically similar to primary corneal epithelial dysplasia or corneal intraepithelial neoplasia without a prominent limbal lesion. We report a case of successful treatment of an unusual case of unilateral central corneal epithelial dysmaturation.

Case report

A 67-year-old man visited the ophthalmology department for an opaque corneal opacity in his right eye, which had developed 1 year before and had gradually increased in size. Slit-lamp examination showed an individual island of opalescent corneal epithelial lesion, 2 × 2.4 mm in size, located in the central cornea, without neoplastic fibrovascular corneal pannus (Figure 1). Best-corrected visual acuity was 20/40 in the right eye and 20/20 in the left eye. Other ocular findings were normal. A decision was made to remove the lesion and subject it to cytological examination. The lesion was carefully removed by simple superficial excision with a knife. Cytological examination of the removed lesion showed normal nuclear/cytoplasmic ratio and hyperplastic epithelium with no atypia (Figure 2). Corneal epithelial dysmaturation was diagnosed based upon the clinical and cytological results. Topical antibiotic and steroid were used for 4 weeks with tapering of the steroid. Visual acuity improved to 20/20 in the right eye. After 3 years of treatment, the lesion had completely regressed and did not recur per slit-lamp examination (Figure 3).

Figure 1
figure 1

Right anterior segment view at the initial examination. It shows an individual island of opalescent corneal epithelial lesion, 2 × 2.4 mm in size, located at central cornea, without neoplastic fibrovascular corneal pannus.

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Figure 2
figure 2

(a) Histopathology of the excised corneal tissue shows increased epithelial cells without surface maturation. There is no inflammatory reaction within the epithelial or stromal layers (haematoxylin–eosin staining; × 40 magnification). (b) Histopathology of the excised corneal tissue reveals a slight loss of polarity of the epithelial cells; however, neither cellular atypia nor dyskeratosis is noted (haematoxylin–eosin staining; × 100 magnification).

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Figure 3
figure 3

Right anterior segment view at the last visit. At 3 years after treatment, the lesion had completely regressed and showed no sign of recurrence.

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Comment

Corneal epithelial dysmaturation tends to exclusively involve the corneal epithelium or involved it in an amount disproportionately large compared with the conjunctiva or limbus.1, 2 Unilateral central corneal epithelial dysmaturation is an extremely unusual group of conditions, which are characterized by the involvement of corneal epithelium only or a disproportionate involvement of the corneal epithelium from a small limbal lesion that has preferentially spread towards the cornea. Epithelial dysmaturation is a benign and indolent process. Lesions may be unilateral or bilateral, stationary or slowly progressive, and may wax and wane spontaneously. Sometimes the opalescent islands and fingers spread over the corneal epithelium and can be mapped geographically over the course of months to years.1 Fortunately, these lesions are indolent and can be treated with simple corneal scraping and, if needed, wide excision of limbal components as with CIN. We report an unusual case of corneal epithelial dysmaturation. It appears that simple superficial excision may be an effective treatment for corneal epithelial dysmaturation.