•This is the first report of aseptic meningitis due to immune checkpoint inhibitor treatment in endometrial cancer.•The meningitis was severe and relapsed multiple times unlike in other reported cases.•Oncologic outcome was excellent after overcoming this severe adverse event.
Keywords: Endometrial cancer; Immune checkpoint inhibitor; Immune-related adverse events; Meningitis; Pembrolizumab.
© 2021 The Authors.