In the present study we report good outcome of oral ketoconazole in patients who were laboratory confirmed to be suffering from Acanthamoeba keratitis. The drug oral route, convenient frequency of doses, minimal side effects and availability at reasonable price, were important factors for selecting ketoconazole. This study involved 138 patients with chronic keratitis who were seen over a 2-year period. Six (4.3%) of these patients were confirmed to be suffering from Acanthamoeba keratitis. Response to ketoconazole was good in four patients at monthly follow up for two years period. Two patients showed initial good ketoconazole response, but had not reported at visits for follow up.