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Oxf Med Case Reports
Oxf Med Case Reports
omcr
Oxford Medical Case Reports
2053-8855
Oxford University Press

10.1093/omcr/omae105
omae105
Case Report
AcademicSubjects/MED00010
omcrep/1600
Secondary corneal keloid: a report of two cases
Sharifi Ali Clinical Research Development Unit, Shafa Hospital, Shafa St. 7616913555, Kerman, Iran

Mirshekari Touraj-Reza Clinical Research Development Unit, Shafa Hospital, Shafa St. 7616913555, Kerman, Iran

Azh Milad Clinical Research Development Unit, Shafa Hospital, Shafa St. 7616913555, Kerman, Iran

Sadeghi Hamid Clinical Research Development Unit, Shafa Hospital, Shafa St. 7616913555, Kerman, Iran

Akbari Zahra Faculty of Medicine, Kerman University of Medical Sciences, Haft-bagh Highway 7616913555, Kerman, Iran

https://orcid.org/0000-0003-4423-4979
Zand Amin Clinical Research Development Unit, Shafa Hospital, Shafa St. 7616913555, Kerman, Iran

Corresponding author. Clinical Research Development Unit, Shafa Hospital, Shafa St. 7616913555, Kerman, Iran. E-mail: sandpost3@gmail.com
9 2024
07 9 2024
07 9 2024
2024 9 omae10505 10 2023
10 1 2024
17 7 2024
© The Author(s) 2024. Published by Oxford University Press.
2024
https://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

Abstract

We conducted clinical and histological evaluations on two male patients who presented with corneal keloid. One patient had a history of corneal trauma due to contact with boiling sunflower oil, while the other had undergone pterygium removal. Upon slit lamp examination, the corneal lesions were identified as single, well-circumscribed, pearly white nodules with a smooth surface. We successfully removed these nodules using a combination of superficial keratectomy and the application of mitomycin C. Light microscopy analysis of the excised nodules revealed hyperplastic epithelium, disrupted Bowman’s layer, and irregularly arranged abundant collagen fibers within the stroma. Notably, there was no recurrence of the lesions in either case within six months following the surgical excision. Secondary corneal keloids should be considered as a potential diagnosis in patients with elevated corneal nodules, especially when there is a history of ocular surface trauma or surgery.

corneal keloid
ocular surface
superficial keratectomy
mitomycin C
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pmcIntroduction

Corneal keloid is a benign fibrous tissue proliferation within the superficial stroma of the cornea. Typically, these lesions manifest as pearly-white, solitary, elevated, and slowly progressing nodules [1]. They are relatively rare, with most cases arising following ocular trauma or surgical interventions on the ocular surface [1]. Primary corneal keloids are exceedingly uncommon, typically occurring in cases associated with congenital conditions such as Lowe’s Syndrome [2–4]. In this report, we present the clinical characteristics, histopathological findings, and surgical outcomes of two cases involving secondary corneal keloids resulting from ocular surface trauma and surgery.

Case reports

This study adhered to the principles outlined in the Declaration of Helsinki, and written informed consent was obtained from both participants.

Case 1

A 69-year-old male presented with a progressive corneal opacity in his left eye over the past six months. He had a history of ocular surface injury caused by splashes of boiling sunflower oil in the affected eye approximately one year ago. Initially, this injury was managed conservatively with topical betamethasone, artificial tears, and a therapeutic contact lens. The patient had no significant medical or family history. At the time of presentation, the best-corrected visual acuity (BCVA) in his right eye was 20/25, while in the left eye, it was 20/80.

On slit-lamp biomicroscopy, a well-defined, pearly-white elevated lesion with a smooth surface was observed in the inferior sector of the left cornea. A few fine stromal vessels were seen entering the lesion (Fig. 1A). Examination of the right cornea was unremarkable. Anterior segment optical coherence tomography (OCT) revealed a well-demarcated lesion located in the subepithelial to anterior corneal stroma, with a clear underlying posterior stroma (Fig. 1B).

Figure 1 (A) Slit-photo of the left cornea displaying a pearly white elevated lesion with a smooth surface and a few stromal vessels. (B) Anterior segment optical coherence tomography (OCT) revealing a high-intensity and well-demarcated lesion in the subepithelial region and the anterior corneal stroma, with a clear underlying posterior stroma. (C) Hematoxylin–eosin staining of the excised lesion showing hyperplastic corneal epithelium (arrows), disruption of Bowman’s layer, and an increase in collagen bundles of the stroma with irregular arrangements (arrowheads). (D) On the 7th day after the lesion excision, the cornea appears clear, with only a trace subepithelial haziness remaining.

One week after the initial visit, we performed superficial keratectomy, followed by the dissection of the lesion from the corneal stroma. Subsequently, 0.02% mitomycin C (MMC) was applied for one minute. Further, superficial scraping with a surgical blade was performed from the central cornea towards the limbus to remove any remaining fibrous tissues. At the conclusion of the surgery, a therapeutic contact lens was placed over the cornea. During the postoperative period, the patient received topical eye drops of chloramphenicol 0.5% and prednisolone 1% (four times a day), with a tapering schedule over eight weeks.

The resected specimen was fixed in 10% formalin and subjected to histopathological examination. Light microscopy revealed hyperplastic epithelium, the absence of Bowman’s layer, and irregular collagen fibrils in the stroma (Fig. 1C). These clinical and histopathological findings were consistent with a diagnosis of corneal keloid. On postoperative day 7, BCVA improved to 20/20, with the cornea appearing clear except for a trace subepithelial haze (Fig. 1D). BCVA remained stable, and there was no recurrence of the lesion during six months of follow-up.

Case 2

A 44-year-old male presented with a gradually enlarging, pearly-white opacity in the right cornea. The patient had a history of pterygium removal from the nasal canthus of the affected eye, involving an autologous conjunctival graft performed eight years earlier. He denied any history of ocular trauma or systemic inflammatory diseases. The BCVA in the affected eye was initially measured at 20/40.

Slit-lamp biomicroscopy revealed a pearly-white nodule with new vessels adjacent to the limbus in the right cornea (Fig. 2A). The fellow cornea exhibited no abnormalities. The lesion was excised using a combination of superficial keratectomy and 0.02% MMC, following the same technique as described in the previous case. Histopathological examination confirmed the presence of a hyperplastic epithelial layer, the absence of Bowman’s layer, and abundant collagen fibers in the anterior stroma (Fig. 2B), consistent with a diagnosis of corneal keloid.

Figure 2 (A) Slit-photo of the ocular surface of the right eye illustrating a pearly white nodule with new vessels in the right cornea. (B) Hematoxylin–eosin staining revealing a hyperplastic epithelial layer (arrows), the absence of Bowman’s layer, and abundant collagen fibers in the anterior stroma (arrowheads). (C) Seven days after the surgery, the cornea is nearly clear.

One week post-surgery, the patient’s BCVA improved to 20/25, and the cornea was nearly clear (Fig. 2C). This improvement persisted through the six-month follow-up period.

Discussion

Corneal keloids are characterized by hyperplastic epithelium, the absence of Bowman’s membrane, irregular and abundant collagen bundles, and a massive proliferation of fibroblast cells [1]. These lesions may also present with new superficial vessels [1, 5]. This disorder is rare and exhibits a higher prevalence among males [1, 2].

In this report, we describe two male patients with unilateral corneal keloids that developed following contact with boiling oil and pterygium removal surgery. The described secondary causes typically occur months to years after the initial event and tend to extend beyond the border of the initial injury, similar to our cases [6, 7]. Primary corneal keloids, on the other hand, are exceedingly rare, tend to be bilateral, and are often associated with congenital diseases such as Lowe’s syndrome [4].

We hypothesize that stromal fibroblasts became hyper-activated following ocular surface trauma or pterygium surgery, leading to the formation of corneal keloids in these cases. Previous reports have indicated that residual fibroblasts persisting at the site of pterygium removal or from the creation of small corneal incisions during cataract surgery may accelerate fibroblast activation [8, 9].

In patients with corneal keloids, recurrence has been reported following various treatments, including SK, phototherapeutic keratectomy, lamellar keratoplasty, and penetrating keratoplasty [2, 10]. Lee et al. reported recurrence of corneal opacification or keloid formation in all four of their cases, between 4 and 10 months after keloid removal [5]. Some investigators have suggested the use of topical MMC during surgery for recurrent corneal keloids, but Lee et al. reported keloid relapse in two cases treated with adjuvant 0.02% MMC during the second surgery [5]. Key factors in the recurrence of corneal keloids include the degree of vascularization, innervation, and myofibroblastic transformation at the site of the removed lesion [2]. Recurrence is more likely in lesions supplied by new vessels originating from the limbus vasculature [5].

In conclusion, secondary corneal keloids should be suspected in patients who present with elevated corneal nodules, especially in cases with a history of ocular surface trauma or surgery. Given the risk of recurrence, long-term follow-up in these patients is essential.

Acknowledgements

The authors are grateful to the research participant and all the hospital staff who took interest and helped out in the study.

Conflict of interest

The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The authors received no financial support for the research, authorship, and/or publication of this article.

Ethical approval

Ethical approval was obtained by the hospital ethics committee.

Consent

Informed patient consent was obtained.

Guarantor

Author Amin Zand is nominated as guarantor of this article.
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