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To review the clinical presentations, risk factors, medical and surgical management, and outcomes of patients with Acanthamoeba keratitis (AK).Retrospective review of laboratory and medical records of all patients suspected of having AK from January 1999 through May 2008 at Cole Eye Institute.Twenty-nine eyes of 26 patients were identified as having either culture- or tissue-proven AK or presumed AK based on clinical examination and complete response to full course of treatment. The most common risk factors identified for AK were history of contact lens wear (89.7%) and exposure to contaminated water (27.6%). Clinical presentations included early AK (superficial disease) in 37.9% of eyes or late AK (deep stromal disease with or without epithelial disease) in 62.1% of eyes. All early AK cases had best-corrected visual acuity of 20/30 or better at last follow-up, whereas only 55.6% of late AK cases achieved 20/30 or better. Eight eyes underwent penetrating keratoplasty. One patient demonstrated viable-appearing cysts in the corneal button, despite 15 months of maximum medical treatment and 5 months off all medical treatments. Over the nearly 10-year period, there was no significant increase in the number of cases seen each year.The most common risk factor for AK continues to be contact lens wear. AK requires prolonged and intense treatment, although good final visual acuity can be achieved. Potentially viable Acanthamoeba cysts can still persist in a noninflamed cornea after extensive medical therapy, supporting the practice that corneal transplantation after presumably resolved cases of AK should be followed with vigilance to detect the earliest signs of recurrent disease.