Our case demonstrated the rare occurrence of autoimmune limbic encephalitis in case of cervical squamous cancer with GABABR, neuronal surface receptor, antibody-mediated CNS inflammation presenting with seizures, encephalopathy status post immunotherapy with pembrolizumab, an immune checkpoint inhibitor.

Anti-GABABR antibody mediated limbic encephalitis is uncommon but potentially treatable condition and should be in differential diagnosis of patients presenting with new-onset seizures, refractory seizures, memory loss, and altered mental status in context of cancer or should be evaluated thoroughly for underlying cancer if presented with limbic encephalitis; early diagnosis and prompt treatment are vital for better outcomes.