Primary leptomeningeal glioblastomatosis is a rare and fatal tumor of the central nervous system, the condition is characterized by diffuse infiltration of the tumor in the meninges without evidence of primary tumor within the brain or spinal cord. We reported an unusual case of leptomengial glioblastomatosis, which was detected by the consecutive cerebrospinal fluid (CSF) cytology with application of immunohistochemistry, in addition to its cytologic findings. A healthy 21 year old man, who was enlisted in the army, presented with a stuporous mental state and diffuse enhancement of meninges without evidence of primary mass lesion in the brain and spinal cord on magnetic resonance imaging(MRI). CSF cytology showed small loose clusters of tumor cells with single cells and lymphocytes. The tumor showed variable pleomorphism with coarse chromatin, irregular nuclear membranes and multi lobated nuclei. On immunohistochemical staining, the tumor cells were founded to be positive for GFAP. In conjunction with radiologic findings, brain biopsy confirmed the diagnosis of leptomenigeal glioblastomatosis. The use of immunohistochemistry is helpful in confirming CSF cytologic diagnosis in patients with primary leptomeningeal glioblastomatosis.