%0 Case Reports %T Case Report: First Successful Treatment of Acanthamoeba Brain Abscess with Combination Surgical Excision and Miltefosine-Led Antimicrobial Therapy. %A Chan A %A Smith S %A Tan E %A Kuruvath S %J Am J Trop Med Hyg %V 106 %N 3 %D 01 2022 17 %M 35026730 %F 3.707 %R 10.4269/ajtmh.21-0166 %X Acanthamoeba brain abscess is very rare and most often fatal. There remains no standardized regimen for its management. We report a case in northern Australia of an immunosuppressed 57-year-old man who presented with diarrhea and weight loss, and was diagnosed with multiple Acanthamoeba brain abscesses after neurological deterioration. This case is the first successful treatment with surgical excision followed by combination antimicrobial therapy including miltefosine. This case was treated initially as nocardiosis or melioidosis, emphasizing the importance of considering differentials such as Acanthamoeba during workup of atypical infection. We present a literature review of the 14 Acanthamoeba brain abscess cases reported in the English literature, of which five were successfully treated. Our review shows a predilection for multiple brain abscesses and an increased mortality rate compared with the general brain abscess population.