2026 Volume 44 Issue 1 Pages 1-6
Synopsis: Immune checkpoint inhibitors have significantly improved the outcomes of uterine malignancies; however, immune-related adverse events can affect multiple organs with diverse clinical manifestations and may be life-threatening. Central nervous system irAEs are rare, with meningitis and encephalitis reported in approximately 0.1-0.2%of cases. We report a case of irAE-associated aseptic meningitis in a patient with recurrent cervical neuroendocrine carcinoma treated with cemiplimab. The patient developed a headache on day 5 after the first dose and presented on day 8 with worsening headache, low-grade fever, and nausea. She was alert, with a positive Jolt accentuation test, but no neck stiffness or Kernig's sign. Brain CT and MRI revealed no abnormalities. Cerebrospinal fluid analysis confirmed aseptic meningitis. Her symptoms rapidly improved with prednisolone at 1 mg/kg/day, and cemiplimab was resumed on day 62 without recurrence. This case highlights the importance of recognizing irAE meningitis, even in the presence of subtle neurological symptoms, and the need for prompt, multidisciplinary management during ICI therapy.Key words: uterine cervix, immune checkpoint inhibitor (ICI), immune-related adverse event (irAE), aseptic meningitis, cemiplimab