Abstract
Background: Gliosarcoma (GS) represents a rare variant of glioblastoma in the central nervous system, characterized by biphasic histopathological features of gliomatous and sarcomatous components. Here, we present an unusual case of GS, which also demonstrated osteosarcomatous component. Case presentations: The patient underwent gross total resection (GTR) of right temporal lobe lesion. Subsequently received external beam radiation therapy with 60 Gy and chemotherapy, postoperatively. The sarcomatous portion of the typical fibrosarcoma pattern mingled with areas of osteoid structure in this 65-year-old feminine case. The molecular pathological analysis demonstrated IDH1/2 wild-type and MGMT promoter island methylated phenotype. Target Enrichment Sequencing (TES) was performed on the gliomatous and sarcomatous components of the tumor tissues. TERT promoter, RB1, NF1, TP53 mutations and copy number variations (CNVs) on chromosome 7, 10q, 11q, 12, 13, 17 and 22 were observed in gliomatous and fibro-sarcomatous mixed tumor tissue; While we found TERT promoter, RB1, TP53 mutations and CNVs on chromosome 2q, 3q, 7, 8, 9, 10, 11, 12, 13, 15, 16, 17, 18, 19 and 22 in osteosarcomatous tumor tissue. Noteworthy, EGFR amplification was not observed in both gliomatous and sarcomatous tumor tissues. Conclusions: In conclusion, integrated with histopathology, molecular pathology, and genomic alteration analysis, we report a case of GS with an extremely rare histopathologic phenotype of osteosarcomatous differentiation, also suffered lung multi-metastases. Additionally, by reviewing the literature, our study of this unusual differentiation of GS into osteosarcoma provides novel insight into the natural history of GS.