Rationale: Astroblastoma is a rare tumor of the central nervous system with uncertain biological behavior and origin. Its histopathological features have been well established, while, to our knowledge, astroblastoma with oligodendroglial-like cells have not been reported.Patient concerns: A 15-year-old girl presented with nausea, vomiting, headache, and visual disturbance.Diagnosis: Magnetic resonance imaging revealed a large neoplasm in the left temporal. Histologically, the tumor showed solid and pseudopapillary structure. Immunohistochemical staining showed that the tumor cells were positive for glial fibrillary acidic protein and vimentin. The oligodendroglial-like cells were positive for glial fibrillary acidic protein, vimentin, and oligodendrocyte transcription factor 2. The antigen KI67 labeling index was about 4%. Sequencing for isocitrate dehydrogenase (IDH) 1 codon 132 and IDH2 codon 172 gene mutations showed negative results. Furthermore, fluorescent analysis revealed neither 1p nor 19q deletion in the lesion. Based on these findings, the girl was finally diagnosed as astroblastoma.Interventions: A craniotomy with total excision of the tumor was performed.Outcomes: The follow-up time was 1 year, no evidence of disease recurrence was found in magnetic resonance imaging.Lessons: Cerebral astroblastoma with oligodendroglial-like cells is a clinically rare tumor of central nervous system. Clear distinction and diagnosis are critical. Abbreviations: EMA = epithelial membrane antigen, FISH = fluorescent in situ hybridization, GFAP = glial fibrillary acidic protein, IDH = isocitrate dehydrogenase, NeuN = neuronal nuclear antigen, Olig-2 = oligodendrocyte transcription factor 2, P53 = tumor protein 53, Syn = synaptophysin.