{Reference Type}: Case Reports {Title}: Case Report: Management of Acanthamoeba Rhinosinusitis in a Patient with Chronic Lymphocytic Leukemia. {Author}: Di Valerio E;Justice JM;Gushiken AC; {Journal}: Am J Trop Med Hyg {Volume}: 111 {Issue}: 1 {Year}: 2024 Jul 3 {Factor}: 3.707 {DOI}: 10.4269/ajtmh.23-0582 {Abstract}: Infections caused by free-living amoebae pose a significant public health threat owing to growing populations of immunocompromised hosts combined with diagnostic delays, treatment difficulties, and high case fatality rates. Nasopharyngeal infections caused by Acanthamoeba are rare and the optimal treatment is not well established. We report a case of Acanthamoeba rhinosinusitis in a patient with chronic lymphocytic leukemia who presented with headaches and chronic rhinosinusitis refractory to multiple courses of antibiotics. A diagnosis of Acanthamoeba rhinosinusitis was established through broad-range polymerase chain reaction testing on sinus tissue. The patient had a favorable response to treatment, which included surgical debridement, cessation of immunosuppressants, and a three-drug regimen consisting of miltefosine, fluconazole, and sulfadiazine.