A healthy 31-year-old female patient developed 2 weeks after delivery a right monocular loss of vision with ocular pain associated with eye movements. She had a right optic disc swelling with right central scotoma and relative afferent pupillary deficit. A prompt clinical recovery was observed after a 3-day course of intravenous methylprednisolone (1 g/day). Cerebrospinal fluid (CSF) examination was normal. Brain magnetic resonance imaging (MRI) showed a right optic nerve edema in its intra-orbital segment (Fig. 1), without any associated white matter lesion. The cervical spine MRI was unremarkable. The right optic neuropathy relapsed 3 weeks later and was again treated with intravenous methylprednisolone (1 g/day, 3 days). The pattern visual evoked potentials latencies were delayed in the right eye (124 ms for the smaller size and 116 ms for the larger size of the checkerboard pattern) and normal in the right eye (106 ms for the smaller size and 99 ms for the larger size of the checkerboard pattern). Ophthalmological examination did not show any persistent optic disc swelling, but revealed a dyschromatopsia and reduced visual acuity at 0.5 in the right eye. Serum TSH, angiotensin converting enzyme, vitamin B12 and folic acid were normal. Anti-viral, anti-borrelia, anti-mycoplasma pneumonia and VDRL were negative. ANA, ANCA, and anti-AQP4 (IgG-NMO) antibodies were absent. Chest-computerized tomography was normal. A third relapse occurred 3 weeks later. She was again given intravenous methylprednisolone (1 g/day, 3 days). This treatment was later relayed by a combination of azathioprine (100 mg/day) and oral methylprednisolone at the initial dose of 48 mg per day. A normalization of the right visual acuity was rapidly obtained. The oral steroids were progressively reduced and discontinued 1 year later, due to a mild cataract. No further relapse was observed after 18 months of evolution.