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Purpose: To define the outcome of acute monosymptomatic optic neuritis (AMON) in Northern Sardinia, Italy. Methods: Records of all AMON patients admitted at the Institute of Ophthalmology, University of Sassari, between 1991 and 2002 were reviewed. The Kaplan-Meier method was used to calculate the cumulative probability of remaining free from clinically definite multiple sclerosis (CDMS). Results: 22 patients (16 women, 6 men) met the inclusion criteria. Mean age at onset was 29.4 years. Mean age-adjusted incidence was 0.91 per 100,000 population per year. Best-corrected visual acuity (BCVA) at presentation was < 20/200 in 12 (54.5%) patients. Brain MRI revealed evidence of two or more demyelinating lesions in all patients. Five (22.7%) eyes had final BCVA < 20/200. Fifteen (68.2%) patients developed CDMS within 7 years. The cumulative probability of remaining free from CDMS was 45% after 36 months and 26% after 82 months. Conclusions: Results suggest that Sardinian patients with AMON may have a worse visual prognosis and a higher risk of developing CDMS.