Background: Spinal meningiomas are relatively uncommon, and meningioma recurrence and malignant transition are even rarer. We report a case of benign spinal meningioma developing into malignancy via cerebrospinal fluid dissemination. Case: A 52-year-old man presented with bilateral lower limb weakness caused by an intradural meningioma (WHO grade I). The meningioma recurred in situ 18 months after the tumor resection and disseminated through cerebrospinal fluid within several months. Cytological testing of cerebrospinal fluid found a multinucleate tumor cell, which helped to confirm the diagnosis of malignancy. Conclusion: We provide a case demonstrating that benign spinal meningiomas can recur and develop into malignancy. Cerebrospinal fluid cytology could be useful as an alternative means of diagnosis when it is difficult to obtain the pathology of a spinal biopsy. © Science Printers and Publishers, Inc.