Purpose: To investigate ocular asymmetry status 5 years after cranioplasty in children with unilateral coronal craniosynostosis. Methods: The medical records of 41 children who underwent cranioplasty at a mean age of 11.7 months were retrospectively reviewed. The cranial vault asymmetry index (CVAI) and the amount of head tilt were measured on images obtained before cranioplasty. Presence of anisometropia, aniso-astigmatism, amblyopia, and strabismus was evaluated at a mean age of 6.8 years. Correlations among the CVAI, direction of synostotic suture and ocular asymmetries were analyzed. Results: Before cranioplasty, the CVAI was 4.5%; 31 of 41 patients (75.6%) exhibited head tilting, which was contralateral to the synostotic suture site in 29 of these patients. At a mean of 5.6 years after cranioplasty, anisometropia of ≥ 1.00 diopters on the spherical equivalent was present in 9 patients (22.0%) and aniso-astigmatism of ≥ 1.00 diopters in 10 patients (24.4%). Amblyopia was in 15 patients (36.6%); the eye contralateral to the synostotic suture was more frequently affected, and anisometropic amblyopia was the most common subtype. Strabismus was present in 28 patients (68.3%); exodeviation and vertical deviation were the most common subtypes. The non-dominant eye was the eye ipsilateral to the synostotic suture in 12 of 13 patients (92.3%) with simulated superior oblique palsy and in 7 of 10 patients (70.0%) with dissociated vertical deviation. In children with CVAI of ≥ 5%, anisometropia was significantly more common than in other children (p = 0.04), but we found no relationship between any other type of ocular asymmetry and a high CVAI. Conclusions Ocular asymmetries including anisometropia, aniso-astigmatism, amblyopia, and strabismus were observed even 5 years after successful cranioplasty treatment for unilateral coronal craniosynostosis, emphasizing the need for continuous ophthalmic follow-up.