BACKGROUND Fungal keratitis is a rare but serious condition that may result in loss of vision. The potentially poor prognosis might be due to a delay in diagnosis and/or to limited treatment options. The aim of this study is to evaluate the clinical outcome of patients treated with topical fluconazole 0.2% for the treatment of filamentous fungal keratitis. METHODS Retrospective case series. A chart review of all patients evaluated at the Ophthalmology Department of the Hospital de Clínicas in Paraguay from January 1997 to December 2000 identified 25 cases of fungal keratitis. Among these cases, one patient discontinued the treatment and another received amphotericin as the first line drug, resulting in 23 cases available for data analysis. Twelve patients were treated with topical fluconazole 0.2% alone (Group I) and 11 patients received a combination of topical fluconazole 0.2% and oral ketoconazole 200 mg twice daily (Group II). RESULTS Sixteen of 23 cases showed resolution of the keratitis, 9/12 (75%) in Group I and 7/11 (64%) in Group II. Seven patients (30%) did not respond to medical treatment and required a surgical procedure to preserve eye integrity. Superficial and small ulcers have a significantly better prognosis than do lrge and deep ulcers. Concomitant oral ketoconazole (400 mg/day) did not improve the clinical results. CONCLUSIONS Our findings of study suggest that topical fluconazole 0.2% is a safe and effective antifungal drug for the management of filamentous mycotic keratitis, particularly in cases that are not severe. The addition of oral ketoconazole to topical fluconazole did not improve the clinical outcome.