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Carcinoid tumors with a primary site in the central nervous system have not been reported in literature yet. We report here about a 41-year-old patient with recurrent and progressive low back pain and bilateral S1 radiculopathy on admission. The patient underwent hemi-laminectomies of the vertebral bodies L5 and S1 and an en bloc resection of the tumor. Postoperative histopathological examination resulted in a well-differentiated intrathecal neuroendocrine tumor (carcinoid) of the terminal filum. Postoperative staging showed no pathological abnormalities and no tumor recurrence after 6 months. Even though rare, carcinoids should be considered as differential diagnosis of tumors occurring in the CNS.