Rationale:Megalophthalmos anterior is a rare, bilateral, nonprogressive, hereditary, congenital disorder, characterized by the enlargement of all anterior segment structures of the eye, with megalocornea, iris atrophy, and zonular abnormalities commonly found. Usually almost asymptomatic in young patients, with most complaints concerning blurred vision due to the common corneal astigmatism, it might in time lead to several complications including premature cataract formation and pigmentary glaucoma.Patient concerns:This review presents the case of a 47-year old patient referred to our clinic for traumatic cataract surgery, with striking bilateral megalocornea, somehow overlooked during previous ophthalmic examinations in his local outpatient clinic.Diagnosis:We noticed markedly enlarged corneas and deepened anterior chambers of his both eyes, accompanied by intumescent, white cataract of the right eye, and incipient cortical cataract of the left eye. Best corrected visual acuity (BCVA) was counting fingers in the right eye and 20/25 in the left eye. Additional examination revealed multiple abnormalities of the anterior segment, leading to the diagnosis of anterior megalophthalmos. It is astounding the patient remained undiagnosed during previous examinations, with his megalocornea and remarkably deep anterior chamber so apparent.Interventions and outcomes:We performed standard phacoemulsification procedure, with 3 piece posterior chamber intraocular lens (PCIOL) implantation into the lens capsule. The surgery was uneventful, with postoperative BCVA of 20/20 in the right eye, and no dislocation of the lens in 9-month observation period.Lessons:Complicated cataract in patients with anterior megalophthalmos can be successfully treated with standard phacoemulsification procedure followed by the bag PCIOL implantation.Care needs to be taken during cataract surgery in these patients, as zonular abnormalities and lens enlargement are common, resulting in increased rate of intra- and postoperative complications. As patients with anterior megalophthalmos require a more careful follow-up.