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Anterior segment dysgenesis (ASD) is a spectrum of disorders affecting the anterior chamber structures and is frequently associated with glaucoma and corneal opacity. It usually has bilateral presentation. We report a case of a 22-year-old male with unilateral Peters anomaly. However, on detailed examination, trabeculodysgenesis was noted in the fellow eye. This highlights the importance of a complete bilateral ocular examination with gonioscopy even in unilateral congenital ASD, which can aid in early diagnosis and management.

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