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The authors describe the unusual case of a patient requiring reoperation for ependymoma of the filum terminale after a symptom-free period of 42 years: treatment of the primary tumor consisted of macroscopically complete surgical excision and postoperative radiotherapy. This case brings to light some interesting observations: a late recurrence, as in the case reported here, appears to be rare especially if the tumor presents in adult age; the long preoperative clinical history (26 months) and the initial aspect of the tumor that infiltrated 2 caudal roots, removed together with the tumor, may have been factors correlated with the risk of recurrence: postoperative radiotherapy may have helped to delay clinical manifestation of the recurrence. Ependymomas of the filum terminale may clinically recur even after complete removal. The latest clinical recurrence described in the literature occurred after 26 years.