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First Hospital of Jilin University, Department of Neurosurgery, Changchun, China DOI : 10.5137/1019-5149.JTN.3961-10.1 The coexistence of spinal teratoma of the conus medullaris and arteriovenous malformation (AVM) is exceptional, which has not been reported previously in the literature. The precise mechanism of the coexistence of these conditions is not known, however, the dysembryonic origin of spinal cord teratoma and AVM seems to play a part in this process. A 34-year-old male patient was admitted with lower back pain, bilateral lower extremity numbness and weakness, and sexual disturbance. Magnetic resonance imaging (MRI) showed an AVM extended cranially from the top of a heterogeneous expansile lesion of the conus medullaris. Surgical exploration and histopathological examination revealed a mature teratoma associated with the AVM. A literature review supported the dysembryonic origin of spinal cord teratomas and AVMs. This unique case may provide insight into the etiopathogenesis of the coexistence of spinal teratoma of the conus medullaris and AVM. Keywords : Spinal teratoma, AVM, Conus medullaris