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Rationale:  Limbal dermoids are choristomas known as congenital benign tumors found in abnormal locations. Despite the benign nature, enlarging limbal dermoids may cause visual abnormalities by cornea infiltration with fat component, visual axis invasion, gradually induced corneal astigmatism, and finally result in anisometropic amblyopia. Here we report a rare case of progressive, large pediatric corneal limbal dermoid in a newborn, managed with tissue glue-assisted monolayer amniotic membrane transplantation.    Patient concerns:  A 1-day-old male baby (gestational age, 36 ± 6 weeks; birth body weight, 2785 gram) presented to our clinic with a whitish mass on his right eye since birth.    Diagnosis:  Ocular examination revealed a solid, whitish-yellow, and ovoid mass with central keratinized epithelium over the superior limbus; the lesion covered two-thirds of the cornea with rapid progression in size. The final pathological examination revealed that the lesion is composed of keratotic lining squamous epithelium resembling epidermis, underling dermal fibrotic connective tissue, and mature fat.    Interventions:  The patient underwent deep lamellar excision followed by mitomycin C (MMC) soaking (0.2 mg/mL, 3 minutes) and tissue glue-assisted monolayer amniotic membrane transplantation with the ring conformer at 2 months of age.    Outcomes:  The ring conformer was smoothly removed 2 weeks after the operation. The patient showed a smooth healing process with less pain and rapid corneal re-epithelization. The ocular surface was stable during the follow-up visits, and no complications were detected. Only mild post-operative scarring over the incision wound was observed.    Lessons:  Although a combination of excision, lamellar keratoplasty, and multilayer amniotic membrane and limbal stem cell transplantation is advocated for the treatment of grade II and III pediatric corneal limbal dermoids, the procedure used in this study offers an alternative surgical approach. However, because of the large size of the lesion and the young age of the patient, the management of amblyopia with visual rehabilitation and corneal transplantation is still needed in the future.

Progressive large pediatric corneal limbal dermoid management with tissue glue-assisted monolayer amniotic membrane transplantation