Background: Peters anomaly is a rare form of anterior segment dysgenesis in which abnormal cleavage of the anterior chamber occurs at the end of the third week of gestation. We examined the prevalence of strabismus and amblyopia and analyzed predictive factors for their development, as well as the visual outcome and associated anomalies in patients with bilateral Peters anomaly. Methods: Using a retrospective review, we identified 25 consecutive patients with bilateral Peters anomaly who were observed between August 1995 and February 2005. Ocular structural and systemic anomalies, amblyopia therapy, visual acuity, and binocular alignment at last visit were recorded. Fisher's exact test was used to identify any association between defined predictive factors and the development of strabismus. Results: Mean follow-up time was 5.1 year (range, 0.5-21 years). Median age at presentation was 2.5 months (range, 1 day to 13 years). Penetrating keratoplasties were performed on 34 eyes in 20 patients. Final best-corrected visual acuity ranged from 20/25 to no light perception. Thirteen of 18 patients with recorded motility (72%) developed strabismus: esotropia (n = 7), exotropia (n = 5), and variable (n = 1); one also had dissociated vertical deviation. Patients with equal vision were either orthophoric (n = 4) or had intermittent esotropia (n = 1), whereas strabismus occurred in 100% of patients whose vision was asymmetric by more than 1.5 octaves. Asymmetric vision was the only statistically significant predictive factor for the development of strabismus (P = 0.002). Amblyopia treatment resulted in improved vision in 3 of 5 patients. Conclusion: Strabismus occurs frequently in bilateral Peters anomaly. Asymmetric vision, (because of ocular structural anomalies) postoperative complications, and amblyopia may predispose to strabismus. Despite ocular structural limitations, amblyopia therapy is recommended in the aggressive rehabilitation of these eyes.