To report a case of choroidal osteoma (CO) complicated by extensive subretinal hemorrhage treated with intravitreal bevacizumab and aflibercept injections.

A 42-year-old female patient presented with decreased visual acuity and a temporal visual field defect in the left eye. The patient had a history of retinal hemorrhage in the left eye 3 years prior, which improved without any treatment. The patient's visual acuity had decreased to 0.6 at the initial visit. On fundus examination, orange-colored elevated lesions involving the superior peripapillary area with massive subretinal hemorrhage extending to the macular area were revealed. Optical coherence tomography, fluorescein angiography, and B-scan ultrasonography results indicated CO complicated by choroidal neovascularization (CNV). With multiple intravitreal injections of bevacizumab and aflibercept (bevacizumab ×1, aflibercept ×2), the patient's visual acuity improved and the CNV lesion was kept stable without recurrence as of the 1-year follow-up visit.

Intravitreal bevacizumab and aflibercept injections can be helpful in the treatment of CO complicated by CNV, by improving visual acuity and the retinal anatomy.