To describe clinical and imaging findings in a young boy presenting with bilateral rapidly progressive necrotizing macular retinitis. A 12-year-old Asian Indian boy developed bilateral progressive macular retinitis. He had generalized tonic-clonic seizures for the past 3 months and gave a history of poor scholastic performance with dementia of recent onset. Multimodal imaging comprising and detailed systemic and laboratory work-up was performed. Both eyes showed rapidly progressive full-thickness retinitis lesions observed as disruption of retinal architecture in both eyes. Left eye optical coherence tomography shows full-thickness retinal involvement with sparing of the internal limiting membrane. Electroencephalogram and magnetic resonance imaging (brain) were suggestive of subacute sclerosing panencephalitis and the diagnosis was confirmed by elevated cerebrospinal fluid and serum IgG measles. The patient did not survive despite treatment with systemic interferon therapy. It is important to look for the measles virus as a probable cause of necrotizing retinitis and neurologic symptoms in immunocompetent unvaccinated young patients. Early referral to a neurologist may assist in the early diagnosis of subacute sclerosing panencephalitis and targeted therapy.