Choroidal osteoma is a rare, classically unilateral tumor. Less than half of the cases are complicated by the onset of choroidal neovascularization (CNV). The authors describe a case of bilateral choroidal osteoma in a 10-year-old female patient complicated by the onset of bilateral CNV. The diagnosis was made by multimodal imaging and ultrasonography. Best-corrected visual acuity (BCVA) was 20/63 for the right eye (OD) and 20/25 for the left eye (OS). Interestingly, CNV OD resulted exudative, whereas no signs of fluid were detected OS. OCT angiography (OCTA) clearly detected the presence of CNV in both eyes. The patient underwent a single 1.0 mg/0.025 mL aflibercept injection OD. At follow-up, BCVA improved to 20/32 OD after 1 month and to 20/25 after 3 months, with no exudation recurrence. Both BCVA and imaging findings were unremarkable OS, thus suggesting the possible “quiescent” nature of this CNV.
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