Objective: To report the ocular histopathologic features of a 55-year-old patient with incontinentia pigmenti retinopathy. Design: Observational case report. Participant: A 55-year-old patient with incontinentia pigmenti retinopathy. Methods: Examination of eyes by light microscopy and retinal trypsin digestion. Main Outcome Measures: Clinical and histopathological findings. Results: Histopathologic examination disclosed inner retinal ischemic atrophy, capillary beading, arteriolar-venous anastomoses, preretinal neovascularization, vasculopathy located at the junction of central vascular and peripheral avascular retina, retinal tears, and tractional retinoschisis. Conclusions: Patients with retinal manifestations of incontinentia pigmenti may progress to proliferative vitreoretinopathy or retinal detachment and should be observed closely over the course of their lifetime.
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