Gliomatosis cerebri (GC) are extensively infiltrative glial tumors classified as astrocytic tumors in the current World Health Organization (WHO) classification scheme. In investigating the tumorigenesis of GC, we reviewed 28 cases of GC immunohistochemically and carried out array-based comparative genomic hybridization (CGH) in ten of those cases. In histological and immunohistochemical review, 18 cases (64%) were of astrocytic lineage, three (11%) were of oligodendroglial lineage, and seven (25%) were of uncommitted lineage. In the array-based CGH, 52 clones were frequently lost on 6p, 9p, and 9q in more than six cases, and six clones were frequently gained on 1p, 6q, 10q, and 21q in more than three cases. Some gained or lost genetic loci differed significantly between the histologically high-grade and low-grade GC (P < 0.05), but unsupervised hierarchical clustering failed to produce evidence of any clinical significance. These altered genetic foci are not known to be involved in the development of conventional glial tumors, including astrocytic tumors. In conclusion, despite the dominant astrocytic differentiation of GC histologically, novel genomic aberrations found in our GC cases were different from those of astrocytic tumors.