Ocular immunology and inflammation · 2025
Remdesivir Treatment Outcomes of Subacute Sclerosing Panencephalitis Presenting with Macular Necrotizing Retinitis: A Case Report.
Клинический случайСтатьяНазвание и аннотация: Перевод в очереди
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Gebara Aseel, Homeidat Mohammed, Keadan Tarek, Fisher-Negev Tamar, Oiknine-Djian Esther, Wolf Dana G, Mechoulam Hadas, Ekstein Dana, Amer Radgonde
Аннотация
PURPOSE: Subacute sclerosing panencephalitis (SSPE) is a fatally progressive neurodegenerative disorder caused by persistent measles virus infection of the CNS. We report on the visual and neurological outcomes of SSPE in a patient who presented with necrotizing retinitis three decades after measles infection.
METHODS: Descriptive case report.
RESULTS: A 31-year-old woman presented with sudden blurred vision of the right eye (RE). Visual acuity (VA) was finger counting in RE and 0.8 in the left eye (LE). Funduscopy revealed RE multifocal macular yellowish lesions, LE peripheral retinal atrophic scar and bilateral optic disc edema. OCT of RE showed "moth-eaten" macula. Elevated anti-measles IgG and positive oligoclonal bands were detected in the cerebrospinal fluid (CSF). Aqueous PCR was positive for Measles. Brain MRI showed mild hyperintense FLAIR signal in the parieto-occipital white matter. Electroencephalogram revealed mild generalized and left temporal focal slowing. Treatment with remdesivir was instituted in combination with intravenous immunoglobulins. Subsequently, intrathecal interferon-ɑ was administered. Macular necrosis progressed relentlessly. Remdesivir lowered measles IgG titer in CSF and serum and the titer re-increased once remdesivir was discontinued. The patient was well one-year later.
CONCLUSION: While remdesivir failed to arrest measles-associated macular necrosis, it may have prevented disease progression in the contralateral eye and in the CNS. To our knowledge, this is the first case of measles-associated necrotizing retinitis that was confirmed by a positive aqueous PCR. In the absence of an established treatment, vaccination remains the most effective modality. Further investigation of remdesivir as a potential treatment of SSPE is required.
METHODS: Descriptive case report.
RESULTS: A 31-year-old woman presented with sudden blurred vision of the right eye (RE). Visual acuity (VA) was finger counting in RE and 0.8 in the left eye (LE). Funduscopy revealed RE multifocal macular yellowish lesions, LE peripheral retinal atrophic scar and bilateral optic disc edema. OCT of RE showed "moth-eaten" macula. Elevated anti-measles IgG and positive oligoclonal bands were detected in the cerebrospinal fluid (CSF). Aqueous PCR was positive for Measles. Brain MRI showed mild hyperintense FLAIR signal in the parieto-occipital white matter. Electroencephalogram revealed mild generalized and left temporal focal slowing. Treatment with remdesivir was instituted in combination with intravenous immunoglobulins. Subsequently, intrathecal interferon-ɑ was administered. Macular necrosis progressed relentlessly. Remdesivir lowered measles IgG titer in CSF and serum and the titer re-increased once remdesivir was discontinued. The patient was well one-year later.
CONCLUSION: While remdesivir failed to arrest measles-associated macular necrosis, it may have prevented disease progression in the contralateral eye and in the CNS. To our knowledge, this is the first case of measles-associated necrotizing retinitis that was confirmed by a positive aqueous PCR. In the absence of an established treatment, vaccination remains the most effective modality. Further investigation of remdesivir as a potential treatment of SSPE is required.