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IMMUNE RECONSTITUTION INFLAMMATORY SYNDROME CAUSING PROGRESSIVE OPTIC NERVE EDEMA IN CRYPTOCOCCAL MENINGITIS

Werner, Astrid C. MD*; Vuong, Laurel N. MD; Hedges, Thomas R. MD; Baumal, Caroline R. MD

Retinal Cases and Brief Reports: Summer 2019 - Volume 13 - Issue 3 - p 207–210
doi: 10.1097/ICB.0000000000000582
Case Report
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Purpose: To report an human immunodeficiency virus–positive patient undergoing therapy for cryptococcal meningitis who developed progressive optic disk edema that was steroid responsive.

Methods: Observational case report.

Results: One month after commencing antifungal treatment for cryptococcal meningitis, the patient developed bilateral, progressive, recurrent optic disk edema with subretinal fluid that coincided with initiation of highly active antiretroviral therapy and recovery of CD4+ cell counts. Lumbar puncture revealed normal opening pressure, and cerebrospinal fluid showed no recurrence of cryptococcal infection. There was no evidence of uveitis. The patient rapidly improved with a 5-day course of high-dose intravenous methylprednisolone.

Conclusion: Recurrent optic disk edema with loss of vision after treatment of cryptococcal meningitis in the setting of normal intracranial pressure may represent a unique manifestation of immune reconstitution inflammatory syndrome localized to the optic nerve without uveitis. This is consistent with the temporal relationship between starting highly active antiretroviral therapy, CD4+ count recovery, and the development of progressive disk edema in the study patient. Isolated optic nerve inflammation as a manifestation of immune reconstitution inflammatory syndrome has not been widely reported.

Vision loss can occur in cryptococcal meningitis, usually because of papilledema. The authors describe a patient with progressive optic disk edema after initiation of highly active antiretroviral therapy in the setting of normal intracranial pressure. This clinical course is consistent with immune reconstitution inflammatory syndrome isolated to the optic nerve.

*Department of Ophthalmology, Tufts New England Eye Center, Boston, Massachusetts;

Neuro-Ophthalmology Service, Department of Ophthalmology, Tufts New England Eye Center, Boston, Massachusetts; and

Retina Service, Department of Ophthalmology, Tufts New England Eye Center, Boston, Massachusetts.

Reprint requests: Caroline R. Baumal, MD, Department of Ophthalmology, Tufts University School of Medicine, New England Eye Center, 260 Tremont Street, Floor 10, Boston, MA 02111; e-mail: cbaumal@gmail.com

Supported by Massachusetts Lions Eye Research Fund and Research to Prevent Blindness.

C. R. Baumal is a speaker and served on an advisory Board for Allergan and Genentech in 2016. The remaining authors have no any financial/conflicting interests to disclose.

© 2019 by Ophthalmic Communications Society, Inc.