Case ReportsA Rare Complication of Cytomegalovirus Infection Meningoventriculoencephalitis Immune Reconstitution Inflammatory SyndromeKerkhoff, Andrew D. MSc*†; Reyes, Juan A. MD, MPH‡; Roberts, Afsoon D. MD*; Simon, Gary L. MD, PhD*Author Information From the *Division of Infectious Diseases, Department of Medicine, The George Washington University School of Medicine and Health Sciences, Washington, DC; †Department of Global Health, Academic Medical Center, Amsterdam Institute for Global Health and Development, University of Amsterdam, Amsterdam, the Netherlands; ‡Division of Hospital Medicine, Department of Medicine, The George Washington University School of Medicine and Health Sciences, Washington, DC. Correspondence to: Andrew D. Kerkhoff, MSc, Division of Infectious Diseases, Department of Medicine, The George Washington University School of Medicine and Health Sciences, 900 23rd St, NW, Washington, DC 20037. E-mail: [email protected]. The authors have no conflicts of interest to disclose. Infectious Diseases in Clinical Practice: November 2014 - Volume 22 - Issue 6 - p 365-367 doi: 10.1097/IPC.0000000000000195 Buy Metrics Abstract Immune recovery uveitis is a type of immune reconstitution inflammatory syndrome (IRIS) associated with cytomegalovirus (CMV) that is well described, however there are very few reports of extraocular CMV–associated central nervous system IRIS clinical manifestations. A 35-year-old human immunodeficiency virus–infected woman receiving antiretroviral therapy for 7 weeks and ganciclovir for 3 months for CMV retinitis presented with a 1-week history of varied neurologic complaints. Since initiating the antiretroviral therapy, she had more than a 4-fold increase in CD4+ count and a 1-log reduction in human immunodeficiency virus viral load. Magnetic resonance imaging showed enhancement of the ventricles and leptomeninges. Cytomegalovirus DNA polymerase chain reaction confirmed the presence of CMV in the cerebrospinal fluid, consistent with a diagnosis of CMV meningoventriculoencephalitis IRIS. Extraocular CMV–associated central nervous system IRIS is exceedingly rare and its clinical presentation seems variable. This report demonstrates successful management with the addition of corticosteroids to anti-CMV therapy with prompt neurological improvement. © 2014 by Lippincott Williams & Wilkins.