Purpose: Congenital corneal leukoma is rare with an incidence of 6/100000 and is one of the most important causes of amblyopia. Here we report an unusual case of bilateral congenital elevated corneal leukoma.

Methods: We encountered a 13-day-old full term male newborn. Ocular examination of both eyes revealed central elevated corneal leukomas with a narrow normal lucid interval to limbus. Corneal diameter of both eyes was 10.5 mm.

Results: After performing penetrating keratoplasty, histological examination reported that corneal thickness was about 2 mm and had central circular defect in Descemet’s membrane, endothelium, and some part of Bowman’s layer compatible with Peters’ anomaly.

Conclusion: Peters’ anomaly should be considered in the differential diagnosis of bilateral congenital bulged corneal leukoma with severe edema.