Adult brainstem gliomas belong to a rare and heterogeneous group of brain tumors. The overall prognosis is poor; therapeutic options are limited, given the resistance to radiotherapy and the unclear role of chemotherapy/antiangiogenic therapy. Apatinib, a tyrosine kinase inhibitor that selectively inhibits the vascular endothelial growth factor receptor and mildly inhibits c-Kit, PDGFR-β, RET, and c-SRC, has been reported to show efficacy among some patients with malignant supratentorial gliomas. However, its effect on brainstem glioma has not been reported so far. Herein, a 66-year-old man with brainstem anaplastic astrocytoma isocitrate dehydrogenase (IDH) wild type was treated initially with combined radiotherapy, temozolomide, and apatinib. The patient achieved a complete response by MRI and continues to have an ongoing progression-free survival of over 8 months. To our knowledge, this is the first case report using apatinib to treat brainstem IDH wild-type anaplastic astrocytoma, displaying an excellent outcome. We also summarize cases of adult brainstem glioma treated with antiangiogenic therapy. Experiences using various regimens may improve understanding of this rare disease, and thus help physicians to seek more effective treatments for these patients.