To report a rare case of neonatal onset multisystem inflammatory disease with serial photographs to characterize the optic nerve findings. Observational case report. A 6 year old girl with neonatal onset multisystem inflammatory disea se, who had received systemic corticosteroid therapy for 5 years, had bilateral fibrillar opacities that surrounded the optic disks and extended into the peripa pillary nerve fiber layer and vessels. A magnetic resonance imaging examination and lumbar puncture revealed elevated intracranial pressure. The elevated intrac ranial pressure returned to normal following a corticosteroid taper. Optic disk photographs, taken 4 years earlier, were subsequently obtained. The optic disk a ppearance had remained unchanged over the 4 year period, consistent with a pseu dopapilledema. The optic disk appearance is not consistent with papilledema from increased intracranial pressure. The optic disk findings, in conjunction with t he underlying inflammatory syndrome, suggest an infiltrative etiology for the at ypical optic nerve findings in neonatal onset multisystem inflammatory disease.
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