A 22-year-old man developed a central scotoma in the left eye during an episode of severe headache 6 months previously. He had a diagnosis of migraine headaches with aura since age 15. Ophthalmic examination showed retinal nerve fiber layer thickness reduction on the papilomacular bundle in the left eye and unremarkable in the right. Optical coherence tomography documented severe macular thickness reduction and a mild and localized retinal nerve fiber layer loss on the temporal side of the optic disc in the left eye. Systemic investigation, neuroimaging and cardiovascular studies were non-revealing. The cause of his visual loss was possibly related to retinal migraine, although an ischemic episode of another origin could not be excluded. Our case is interesting because it draws attention to the fact that optical coherence tomography may be able to identify localized neural loss not only by measuring peripapillary retinal nerve fiber layer but also by means of macular thickness measurements.