Rare giant corneal keloid presenting 26 years after trauma: A case report

Abstract

Background: Corneal keloid is a rare clinical disease with an unknown etiology, which is easily misdiagnosed. Surgery is the most effective treatment but is rarely reported in the literature. Herein, we report the clinical features, histopathology, and surgical outcome of a giant corneal keloid with trophoblastic vessels and discuss the genesis of the mass.

Case summary: A 36-year-old young man was admitted to the hospital because of a large mass on the surface of the left cornea. The patient had suffered an injury to his left eye at the age of 6-years-old; however, as the injury did not cause cornea perforation, he did not undergo treatment. Slit lamp exam showed a large, elevated, opaque lesion that covered the entire cornea and protruded from the surface of the eyeball. Anterior segment optical coherence tomography (AS-OCT) revealed a lesion of irregular density involving the anterior stroma. We suspected a secondary corneal fibroproliferative mass based on the clinical history, and slit lamp and AS-OCT findings. The patient subsequently underwent a superficial keratectomy and keratoplasty, and the final diagnosis of corneal keloid was confirmed by intraoperative histopathological examination.

Conclusion: Non-penetrating corneal trauma damages corneal epithelium basement membrane, initiating stromal fibrosis and causing corneal keloids. AS-OCT and biopsy confirm diagnosis.

Keywords: Anterior segment ocular coherence tomography; Case report; Corneal keloid; Deep anterior lamellar keratoplasty; Histopathology; Immunohistochemical staining.

Figure 1

Preoperative clinical appearance and anterior segment optical coherence tomography images. A and B: Slit lamp microscope images; C: Anterior segment optical coherence tomography images.

Figure 2

Surgery procedure. A: Electrocoagulation of the neovascularization on the surface of the swollen tissue; B-C: The hyperplastic fibrous connective tissue was removed, and the corneal stroma was exposed; D: The temporal nutrient vessels were seen in the stroma, with many branches crossing the pupil; E: The corneal stroma was dissected layer-by-layer until the Descemet’s membrane was approached, and the corneal endothelium was transparent; F: A 9.25-mm implant and implant bed were made, and the two were aligned and sutured.

Figure 3

Histopathological and immunohistochemical results. A-B: Hematoxylin and eosin staining showed the irregular surface of the swelling, non-keratinized epithelium without Bowman’s layer, and dense fibrous connective tissue with blood vessels beneath the epithelium; C: Vimentin staining was diffusely positive within the parenchyma of the mass; D: Smooth muscle actin staining was positive in the smooth muscle walls of the vasculature and myofibroblasts.

Figure 4

Postoperative ocular surface and anterior segment optical coherence tomography images. A: Slit lamp image 1 wk after surgery; B: Slit lamp image 4 mo after surgery; C: Anterior segment optical coherence tomography images 4 mo after surgery.