Background: Peters' anomaly (PA) is a rare form of anterior segment dysgenesis characterized by corneal opacity with varied degree of anterior chamber affection with associated defects in the posterior layers of the cornea.
Case presentation: We report the case of a 3-month-old male infant with bilateral corneal opacity since birth who was transferred from the pediatric unit for further ophthalmic care. He was diagnosed with PA with subsequent systemic-associated malformations including craniofacial defects and skeletal defects. He was managed minimally with topical mydriatics and antiglaucomatous drugs.
Conclusion: PA is an inevitable cause of blindness in developing countries due to poor infrastructure and limited resources.
Keywords: camptodactyly; clinodactyly; congenital corneal opacity; polydactyly.