Abstract
Case Description: 65-year-old female diagnosed with longitudinally extensive transverse myelitis. She had excellent response to immunotherapy despite her atypical features, and her spinal lesion was fully recovered in the second month of the treatment.Findings: Nine months after, she was diagnosed with primary cerebral central nervous system lymphoma while there was no recurrence of the spinal cord lesion as her most recent follow-up visit being 2 years after the LETM.Clinical Relevance: Spinal sentinel neuroinflammation preceding primary central nervous system lymphoma might represent a valuable etiology in the differentials of LETM, as illustrated here.