Giant cell glioblastoma is an extraordinary subtype of glioblastoma that merits mention as a distinct variant of glioblastoma in World Health Organization classification of gliomas. It represents for about 1% of brain tumors and about 5% of glioblastomas in general. It has no preferential region and may occur in various locations of the central nervous system, most commonly in the temporal lobe. To our knowledge, there have been a few cases involved in the unilateral lateral ventricle reported in the literature, but there have been no reports about the recurrent cases involved in bilateral lateral ventricles. We herein present a very rare case of recurrent giant cell glioblastoma that occurred across bilateral lateral ventricles.