Subacute sclerosing panencephalitis (SSPE) is a progressive encephalitis caused by persistent measles infection. The disease is almost always fatal and mainly affects children and young adults. Ophthalmological signs are frequently observed and can be the presenting manifestations. We report the case of a 22-year-old young man who presented with bilateral posterior retinal necrosis confirmed by fluorescein angiography and normal laboratory tests. The appearance of peripheral retinal tears and stereotyped myoclonic movements were signs of the evolving condition. Diagnosis of SSPE was confirmed by brain MRI, EEG, and cerebrospinal fluid examination. The patient's neurological condition deteriorated rapidly and he died 2 months following the onset of ophthalmological manifestations.