Abstract
<title>Abstract</title> <p>Background: complex anterior segment anomalies (nystagmus, micro cornea, acoria, high IOP and iris coloboma) is a rare congenital anomaly characterized by the absence of a pupil, leading to severe visual impairment despite a clear cornea and lens. Pupilloplasty is a therapeutic procedure aimed at restoring a functional visual axis in eyes with iris abnormalities. Visual improvement in children with complex anterior segment anomalies and longstanding visual deprivation is uncommon. Case Presentation: We report a 10-year-old female with bilateral severe visual impairment, horizontal nystagmus, microcornea, and acoria. Preoperative visual acuity was 1/60 in both eyes. Due to the absence of a functional pupil, preoperative evaluation of the lens and posterior segment was not possible. Pupilloplasty was performed in one eye under general anesthesia after identifying an iris coloboma. Postoperatively, visual acuity improved to 6/18 without correction, with resolution of nystagmus and marked functional improvement, allowing the patient to went to school. Conclusion: This case demonstrates that pupilloplasty can serve as a vision-restoring procedure in children with acoria and complex anterior segment anomalies, even at an age when amblyopia treatment is typically less effective.</p>