Abstract
Infantile epileptic spasms syndrome is characterized by epileptic spasms (ES), neurodevelopmental regression, and variable electroclinical features. Early recognition and prompt treatment are crucial for improving outcomes; however, subtle ictal manifestations are often overlooked by caregivers. We describe a male infant who exhibited repeated upward eye deviation beginning at 5 months of age, preceding the onset of typical ES. Prolonged scalp video electroencephalography (EEG) monitoring captured both isolated upward eye deviation and typical ES preceded by upward eye deviation. Despite the introduction of two antiseizure medications, ES persisted. Given the absence of lateralizing findings on neuroimaging and the presence of bilaterally synchronous ictal discharges on EEG, corpus callosotomy (CC) was performed at 7 months of age without prior adrenocorticotropic hormone (ACTH) therapy, in accordance with parental preference. The patient has remained seizure free for 1 year and 3 months postoperatively, with age-appropriate neurodevelopmental scores on the Vineland Adaptive Behavior Scales, Second Edition. This case highlights the importance of identifying subtle signs, such as upward eye deviation, as potential early ictal precursors in ES. CC may represent a viable first-line treatment in selected cases.